Publisher: Sage Publications   (Total: 1166 journals)

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Showing 1 - 200 of 1166 Journals sorted alphabetically
AADE in Practice     Hybrid Journal   (Followers: 6)
Abstracts in Anthropology     Full-text available via subscription   (Followers: 29)
Academic Pathology     Open Access   (Followers: 6)
Accounting History     Hybrid Journal   (Followers: 18, SJR: 0.527, CiteScore: 1)
Acta Radiologica     Hybrid Journal   (Followers: 1, SJR: 0.754, CiteScore: 2)
Acta Radiologica Open     Open Access   (Followers: 2)
Acta Sociologica     Hybrid Journal   (Followers: 39, SJR: 0.939, CiteScore: 2)
Action Research     Hybrid Journal   (Followers: 53, SJR: 0.308, CiteScore: 1)
Active Learning in Higher Education     Hybrid Journal   (Followers: 398, SJR: 1.397, CiteScore: 2)
Adaptive Behavior     Hybrid Journal   (Followers: 9, SJR: 0.288, CiteScore: 1)
Administration & Society     Hybrid Journal   (Followers: 18, SJR: 0.675, CiteScore: 1)
Adoption & Fostering     Hybrid Journal   (Followers: 25, SJR: 0.313, CiteScore: 0)
Adsorption Science & Technology     Open Access   (Followers: 9, SJR: 0.258, CiteScore: 1)
Adult Education Quarterly     Hybrid Journal   (Followers: 262, SJR: 0.566, CiteScore: 2)
Adult Learning     Hybrid Journal   (Followers: 51)
Advances in Dental Research     Hybrid Journal   (Followers: 11, SJR: 1.791, CiteScore: 4)
Advances in Developing Human Resources     Hybrid Journal   (Followers: 35, SJR: 0.614, CiteScore: 2)
Advances in Mechanical Engineering     Open Access   (Followers: 156, SJR: 0.272, CiteScore: 1)
Advances in Methods and Practices in Psychological Science     Full-text available via subscription   (Followers: 20)
Advances in Structural Engineering     Full-text available via subscription   (Followers: 51, SJR: 0.599, CiteScore: 1)
AERA Open     Open Access   (Followers: 14)
Affilia     Hybrid Journal   (Followers: 6, SJR: 0.496, CiteScore: 1)
Africa Spectrum     Open Access   (Followers: 17)
Agrarian South : J. of Political Economy     Hybrid Journal   (Followers: 3)
Air, Soil & Water Research     Open Access   (Followers: 13, SJR: 0.214, CiteScore: 1)
Alexandria : The J. of National and Intl. Library and Information Issues     Full-text available via subscription   (Followers: 68)
Allergy & Rhinology     Open Access   (Followers: 5)
AlterNative : An Intl. J. of Indigenous Peoples     Full-text available via subscription   (Followers: 39, SJR: 0.194, CiteScore: 0)
Alternative Law J.     Hybrid Journal   (Followers: 12, SJR: 0.176, CiteScore: 0)
Alternatives : Global, Local, Political     Hybrid Journal   (Followers: 12, SJR: 0.351, CiteScore: 1)
Alternatives to Laboratory Animals     Full-text available via subscription   (Followers: 11, SJR: 0.297, CiteScore: 1)
American Behavioral Scientist     Hybrid Journal   (Followers: 26, SJR: 0.982, CiteScore: 2)
American Economist     Hybrid Journal   (Followers: 7)
American Educational Research J.     Hybrid Journal   (Followers: 260, SJR: 2.913, CiteScore: 3)
American J. of Alzheimer's Disease and Other Dementias     Hybrid Journal   (Followers: 23, SJR: 0.67, CiteScore: 2)
American J. of Cosmetic Surgery     Hybrid Journal   (Followers: 9)
American J. of Evaluation     Hybrid Journal   (Followers: 18, SJR: 0.646, CiteScore: 2)
American J. of Health Promotion     Hybrid Journal   (Followers: 35, SJR: 0.807, CiteScore: 1)
American J. of Hospice and Palliative Medicine     Hybrid Journal   (Followers: 47, SJR: 0.65, CiteScore: 1)
American J. of Law & Medicine     Full-text available via subscription   (Followers: 12, SJR: 0.204, CiteScore: 1)
American J. of Lifestyle Medicine     Hybrid Journal   (Followers: 7, SJR: 0.431, CiteScore: 1)
American J. of Medical Quality     Hybrid Journal   (Followers: 13, SJR: 0.777, CiteScore: 1)
American J. of Men's Health     Open Access   (Followers: 9, SJR: 0.595, CiteScore: 2)
American J. of Rhinology and Allergy     Hybrid Journal   (Followers: 11, SJR: 0.972, CiteScore: 2)
American J. of Sports Medicine     Hybrid Journal   (Followers: 249, SJR: 3.949, CiteScore: 6)
American Politics Research     Hybrid Journal   (Followers: 36, SJR: 1.313, CiteScore: 1)
American Review of Public Administration     Hybrid Journal   (Followers: 28, SJR: 2.062, CiteScore: 2)
American Sociological Review     Hybrid Journal   (Followers: 358, SJR: 6.333, CiteScore: 6)
American String Teacher     Full-text available via subscription   (Followers: 3)
Analytical Chemistry Insights     Open Access   (Followers: 26, SJR: 0.224, CiteScore: 1)
Angiology     Hybrid Journal   (Followers: 5, SJR: 0.849, CiteScore: 2)
Animation     Hybrid Journal   (Followers: 15, SJR: 0.197, CiteScore: 0)
Annals of Clinical Biochemistry     Hybrid Journal   (Followers: 10, SJR: 0.634, CiteScore: 1)
Annals of Otology, Rhinology & Laryngology     Hybrid Journal   (Followers: 20, SJR: 0.807, CiteScore: 1)
Annals of Pharmacotherapy     Hybrid Journal   (Followers: 59, SJR: 1.096, CiteScore: 2)
Annals of the American Academy of Political and Social Science     Hybrid Journal   (Followers: 51, SJR: 1.225, CiteScore: 3)
Annals of the ICRP     Hybrid Journal   (Followers: 4, SJR: 0.548, CiteScore: 1)
Anthropocene Review     Hybrid Journal   (Followers: 8, SJR: 3.341, CiteScore: 7)
Anthropological Theory     Hybrid Journal   (Followers: 48, SJR: 0.739, CiteScore: 1)
Antitrust Bulletin     Hybrid Journal   (Followers: 14)
Antiviral Chemistry and Chemotherapy     Open Access   (Followers: 2, SJR: 0.635, CiteScore: 2)
Antyajaa : Indian J. of Women and Social Change     Hybrid Journal   (Followers: 1)
Applied Biosafety     Hybrid Journal   (Followers: 1, SJR: 0.131, CiteScore: 0)
Applied Psychological Measurement     Hybrid Journal   (Followers: 21, SJR: 1.17, CiteScore: 1)
Applied Spectroscopy     Full-text available via subscription   (Followers: 27, SJR: 0.489, CiteScore: 2)
Armed Forces & Society     Hybrid Journal   (Followers: 25, SJR: 0.29, CiteScore: 1)
Arthaniti : J. of Economic Theory and Practice     Full-text available via subscription  
Arts and Humanities in Higher Education     Hybrid Journal   (Followers: 49, SJR: 0.305, CiteScore: 1)
Asia Pacific Media Educator     Hybrid Journal   (Followers: 1, SJR: 0.23, CiteScore: 0)
Asia-Pacific J. of Management Research and Innovation     Full-text available via subscription   (Followers: 3)
Asia-Pacific J. of Public Health     Hybrid Journal   (Followers: 15, SJR: 0.558, CiteScore: 1)
Asia-Pacific J. of Rural Development     Hybrid Journal   (Followers: 2)
Asian and Pacific Migration J.     Full-text available via subscription   (Followers: 8, SJR: 0.324, CiteScore: 1)
Asian Cardiovascular and Thoracic Annals     Hybrid Journal   (Followers: 2, SJR: 0.305, CiteScore: 0)
Asian J. of Comparative Politics     Hybrid Journal   (Followers: 5)
Asian J. of Legal Education     Full-text available via subscription   (Followers: 4)
Asian J. of Management Cases     Hybrid Journal   (Followers: 6, SJR: 0.101, CiteScore: 0)
ASN Neuro     Open Access   (Followers: 2, SJR: 1.534, CiteScore: 3)
Assessment     Hybrid Journal   (Followers: 19, SJR: 1.519, CiteScore: 3)
Assessment for Effective Intervention     Hybrid Journal   (Followers: 15, SJR: 0.578, CiteScore: 1)
Australasian J. of Early Childhood     Hybrid Journal   (Followers: 7, SJR: 0.535, CiteScore: 1)
Australasian Psychiatry     Hybrid Journal   (Followers: 18, SJR: 0.433, CiteScore: 1)
Australian & New Zealand J. of Psychiatry     Hybrid Journal   (Followers: 30, SJR: 1.801, CiteScore: 2)
Australian and New Zealand J. of Criminology     Hybrid Journal   (Followers: 547, SJR: 0.612, CiteScore: 1)
Australian J. of Career Development     Hybrid Journal   (Followers: 5)
Australian J. of Education     Hybrid Journal   (Followers: 51, SJR: 0.403, CiteScore: 1)
Australian J. of Management     Hybrid Journal   (Followers: 13, SJR: 0.497, CiteScore: 1)
Autism     Hybrid Journal   (Followers: 358, SJR: 1.739, CiteScore: 4)
Autism & Developmental Language Impairments     Open Access   (Followers: 17)
Avian Biology Research     Hybrid Journal   (Followers: 6, SJR: 0.401, CiteScore: 1)
Behavior Modification     Hybrid Journal   (Followers: 14, SJR: 0.877, CiteScore: 2)
Behavioral and Cognitive Neuroscience Reviews     Hybrid Journal   (Followers: 27)
Behavioral Disorders     Hybrid Journal   (Followers: 2)
Beyond Behavior     Hybrid Journal   (Followers: 2)
Bible Translator     Hybrid Journal   (Followers: 13)
Biblical Theology Bulletin     Hybrid Journal   (Followers: 24, SJR: 0.184, CiteScore: 0)
Big Data & Society     Open Access   (Followers: 55)
Biochemistry Insights     Open Access   (Followers: 7)
Bioinformatics and Biology Insights     Open Access   (Followers: 12, SJR: 1.141, CiteScore: 2)
Biological Research for Nursing     Hybrid Journal   (Followers: 7, SJR: 0.685, CiteScore: 2)
Biomarker Insights     Open Access   (Followers: 1, SJR: 0.81, CiteScore: 2)
Biomarkers in Cancer     Open Access   (Followers: 11)
Biomedical Engineering and Computational Biology     Open Access   (Followers: 14)
Biomedical Informatics Insights     Open Access   (Followers: 8)
Bioscope: South Asian Screen Studies     Hybrid Journal   (Followers: 4, SJR: 0.235, CiteScore: 0)
BMS: Bulletin of Sociological Methodology/Bulletin de Méthodologie Sociologique     Hybrid Journal   (Followers: 4, SJR: 0.226, CiteScore: 0)
Body & Society     Hybrid Journal   (Followers: 29, SJR: 1.531, CiteScore: 3)
Bone and Tissue Regeneration Insights     Open Access   (Followers: 2)
Brain and Neuroscience Advances     Open Access  
Brain Science Advances     Open Access  
Breast Cancer : Basic and Clinical Research     Open Access   (Followers: 12, SJR: 0.823, CiteScore: 2)
British J. of Music Therapy     Hybrid Journal   (Followers: 9)
British J. of Occupational Therapy     Hybrid Journal   (Followers: 253, SJR: 0.323, CiteScore: 1)
British J. of Pain     Hybrid Journal   (Followers: 31, SJR: 0.579, CiteScore: 2)
British J. of Politics and Intl. Relations     Hybrid Journal   (Followers: 39, SJR: 0.91, CiteScore: 2)
British J. of Visual Impairment     Hybrid Journal   (Followers: 14, SJR: 0.337, CiteScore: 1)
British J.ism Review     Hybrid Journal   (Followers: 18)
BRQ Business Review Quarterly     Open Access   (Followers: 1)
Building Acoustics     Hybrid Journal   (Followers: 4, SJR: 0.215, CiteScore: 1)
Building Services Engineering Research & Technology     Hybrid Journal   (Followers: 3, SJR: 0.583, CiteScore: 1)
Bulletin of Science, Technology & Society     Hybrid Journal   (Followers: 9)
Business & Society     Hybrid Journal   (Followers: 15)
Business and Professional Communication Quarterly     Hybrid Journal   (Followers: 9, SJR: 0.348, CiteScore: 1)
Business Information Review     Hybrid Journal   (Followers: 17, SJR: 0.279, CiteScore: 0)
Business Perspectives and Research     Hybrid Journal   (Followers: 3)
Cahiers Élisabéthains     Hybrid Journal   (Followers: 1, SJR: 0.111, CiteScore: 0)
Calcutta Statistical Association Bulletin     Hybrid Journal   (Followers: 1)
California Management Review     Hybrid Journal   (Followers: 37, SJR: 2.209, CiteScore: 4)
Canadian Association of Radiologists J.     Full-text available via subscription   (Followers: 2, SJR: 0.463, CiteScore: 1)
Canadian J. of Kidney Health and Disease     Open Access   (Followers: 8, SJR: 1.007, CiteScore: 2)
Canadian J. of Nursing Research (CJNR)     Hybrid Journal   (Followers: 15)
Canadian J. of Occupational Therapy     Hybrid Journal   (Followers: 168, SJR: 0.626, CiteScore: 1)
Canadian J. of Psychiatry     Hybrid Journal   (Followers: 28, SJR: 1.769, CiteScore: 3)
Canadian J. of School Psychology     Hybrid Journal   (Followers: 12, SJR: 0.266, CiteScore: 1)
Canadian Pharmacists J. / Revue des Pharmaciens du Canada     Hybrid Journal   (Followers: 3, SJR: 0.536, CiteScore: 1)
Cancer Control     Open Access   (Followers: 2)
Cancer Growth and Metastasis     Open Access   (Followers: 1)
Cancer Informatics     Open Access   (Followers: 4, SJR: 0.64, CiteScore: 1)
Capital and Class     Hybrid Journal   (Followers: 10, SJR: 0.282, CiteScore: 1)
Cardiac Cath Lab Director     Full-text available via subscription   (Followers: 1)
Cardiovascular and Thoracic Open     Open Access   (Followers: 1)
Career Development and Transition for Exceptional Individuals     Hybrid Journal   (Followers: 10, SJR: 0.44, CiteScore: 1)
Cartilage     Hybrid Journal   (Followers: 6, SJR: 0.889, CiteScore: 3)
Cell Transplantation     Open Access   (Followers: 5, SJR: 1.023, CiteScore: 3)
Cephalalgia     Hybrid Journal   (Followers: 8, SJR: 1.581, CiteScore: 3)
Cephalalgia Reports     Open Access   (Followers: 4)
Child Language Teaching and Therapy     Hybrid Journal   (Followers: 34, SJR: 0.501, CiteScore: 1)
Child Maltreatment     Hybrid Journal   (Followers: 11, SJR: 1.22, CiteScore: 3)
Child Neurology Open     Open Access   (Followers: 6)
Childhood     Hybrid Journal   (Followers: 19, SJR: 0.894, CiteScore: 2)
Childhood Obesity and Nutrition     Open Access   (Followers: 12)
China Information     Hybrid Journal   (Followers: 9, SJR: 0.767, CiteScore: 2)
China Report     Hybrid Journal   (Followers: 11, SJR: 0.221, CiteScore: 0)
Chinese J. of Sociology     Full-text available via subscription   (Followers: 5)
Christian Education J. : Research on Educational Ministry     Hybrid Journal   (Followers: 1)
Chronic Illness     Hybrid Journal   (Followers: 6, SJR: 0.672, CiteScore: 2)
Chronic Respiratory Disease     Hybrid Journal   (Followers: 12, SJR: 0.808, CiteScore: 2)
Chronic Stress     Open Access  
Citizenship, Social and Economics Education     Full-text available via subscription   (Followers: 6, SJR: 0.145, CiteScore: 0)
Cleft Palate-Craniofacial J.     Hybrid Journal   (Followers: 8, SJR: 0.757, CiteScore: 1)
Clin-Alert     Hybrid Journal   (Followers: 1)
Clinical and Applied Thrombosis/Hemostasis     Open Access   (Followers: 32, SJR: 0.49, CiteScore: 1)
Clinical and Translational Neuroscience     Open Access   (Followers: 1)
Clinical Case Studies     Hybrid Journal   (Followers: 3, SJR: 0.364, CiteScore: 1)
Clinical Child Psychology and Psychiatry     Hybrid Journal   (Followers: 45, SJR: 0.73, CiteScore: 2)
Clinical EEG and Neuroscience     Hybrid Journal   (Followers: 8, SJR: 0.552, CiteScore: 2)
Clinical Ethics     Hybrid Journal   (Followers: 13, SJR: 0.296, CiteScore: 1)
Clinical Medicine Insights : Arthritis and Musculoskeletal Disorders     Open Access   (Followers: 3, SJR: 0.537, CiteScore: 2)
Clinical Medicine Insights : Blood Disorders     Open Access   (Followers: 1, SJR: 0.314, CiteScore: 2)
Clinical Medicine Insights : Cardiology     Open Access   (Followers: 8, SJR: 0.686, CiteScore: 2)
Clinical Medicine Insights : Case Reports     Open Access   (Followers: 1, SJR: 0.283, CiteScore: 1)
Clinical Medicine Insights : Circulatory, Respiratory and Pulmonary Medicine     Open Access   (Followers: 4, SJR: 0.425, CiteScore: 2)
Clinical Medicine Insights : Ear, Nose and Throat     Open Access   (Followers: 2)
Clinical Medicine Insights : Endocrinology and Diabetes     Open Access   (Followers: 34, SJR: 0.63, CiteScore: 2)
Clinical Medicine Insights : Oncology     Open Access   (Followers: 3, SJR: 1.129, CiteScore: 3)
Clinical Medicine Insights : Pediatrics     Open Access   (Followers: 3)
Clinical Medicine Insights : Psychiatry     Open Access   (Followers: 10)
Clinical Medicine Insights : Reproductive Health     Open Access   (Followers: 1, SJR: 0.776, CiteScore: 0)
Clinical Medicine Insights : Therapeutics     Open Access   (Followers: 1, SJR: 0.172, CiteScore: 0)
Clinical Medicine Insights : Trauma and Intensive Medicine     Open Access   (Followers: 4)
Clinical Medicine Insights : Urology     Open Access   (Followers: 3)
Clinical Medicine Insights : Women's Health     Open Access   (Followers: 4)
Clinical Nursing Research     Hybrid Journal   (Followers: 34, SJR: 0.471, CiteScore: 1)
Clinical Pathology     Open Access   (Followers: 5)
Clinical Pediatrics     Hybrid Journal   (Followers: 25, SJR: 0.487, CiteScore: 1)
Clinical Psychological Science     Hybrid Journal   (Followers: 16, SJR: 3.281, CiteScore: 5)
Clinical Rehabilitation     Hybrid Journal   (Followers: 78, SJR: 1.322, CiteScore: 3)
Clinical Risk     Hybrid Journal   (Followers: 5, SJR: 0.133, CiteScore: 0)
Clinical Trials     Hybrid Journal   (Followers: 22, SJR: 2.399, CiteScore: 2)
Clothing and Textiles Research J.     Hybrid Journal   (Followers: 28, SJR: 0.36, CiteScore: 1)
Collections : A J. for Museum and Archives Professionals     Full-text available via subscription   (Followers: 3)
Common Law World Review     Full-text available via subscription   (Followers: 17)
Communication & Sport     Hybrid Journal   (Followers: 8, SJR: 0.385, CiteScore: 1)
Communication and the Public     Hybrid Journal   (Followers: 2)
Communication Disorders Quarterly     Hybrid Journal   (Followers: 15, SJR: 0.458, CiteScore: 1)
Communication Research     Hybrid Journal   (Followers: 24, SJR: 2.171, CiteScore: 3)
Community College Review     Hybrid Journal   (Followers: 8, SJR: 1.451, CiteScore: 1)
Comparative Political Studies     Hybrid Journal   (Followers: 293, SJR: 3.772, CiteScore: 3)
Compensation & Benefits Review     Hybrid Journal   (Followers: 8)
Competition & Change     Hybrid Journal   (Followers: 12, SJR: 0.843, CiteScore: 2)

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Similar Journals
Journal Cover
Cleft Palate-Craniofacial Journal
Journal Prestige (SJR): 0.757
Citation Impact (citeScore): 1
Number of Followers: 8  
 
  Hybrid Journal Hybrid journal (It can contain Open Access articles)
ISSN (Print) 1545-1569 - ISSN (Online) 1055-6656
Published by Sage Publications Homepage  [1166 journals]
  • Interdisciplinary Considerations for Nasolabial Repair During a Global
           Pandemic

    • Free pre-print version: Loading...

      Authors: Pauline Joy F. Santos, Omotayo A. Arowojolu, Raj M. Vyas
      Pages: 1341 - 1347
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1341-1347, November 2021.
      Background:At the declaration of the global pandemic on March 11, 2020, many hospitals and institutions developed a tiered framework for the stratification and prioritization of elective surgery. Cleft lip and palate repair was classified as low acuity, and nasoalveolar molding (NAM) clinics were closed. Anticipating the consequences of delayed cleft care and the additional burden this would cause families, we reassessed our risk-stratification and perioperative algorithms. We hypothesized we could safely optimize nasolabial repair without burdening our care systems and without increasing COVID-19–related morbidity/mortality.Methods:Our multidisciplinary cleft team reevaluated patient selection to maximize surgical impact. Perioperative protocols were adjusted, and COVID-19 preoperative testing was utilized before nasolabial repair and prior to suture removal under anesthesia.Results:Early in the pandemic, unilateral cleft repair was prioritized and successfully completed on 9 patients. There were no complications related to COVID-19. Nasoalveolar molding clinic was reopened after total patient volume was significantly decreased.Conclusions:We offer an approach for surgical management of nasolabial clefts during a global pandemic. Although guidelines have suggested postponing all cleft care, we found that at our dedicated pediatric hospital with low burden of COVID-19 and adequate resources, we could follow a strategy to safely resume cleft care while decreasing burden on our patients’ families and care delivery systems.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-02T09:34:51Z
      DOI: 10.1177/1055665621993970
      Issue No: Vol. 58, No. 11 (2021)
       
  • Furlow Palatoplasty: Should We Also Focus on the Size of the
           Nasopharynx'

    • Free pre-print version: Loading...

      Authors: Jacques E. Leclerc, Francis Gilbert, Élisa-Maude McConnell, Ericka Beaudoin, Johanie Bouchard, David Simonyan
      Pages: 1348 - 1360
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1348-1360, November 2021.
      Objectives:(1) To determine the incidence rate of velopharyngeal dysfunction (VPD) according to 7 speech criteria post-Furlow palatoplasty. (2) To find an anatomical measurement of the cleft palate (or combination of measurements) associated with the occurrence of VPD.Study design:Retrospective cohort study.Participants and Methods:Fifty-six patients with cleft palate ± cleft lip underwent palatoplasty with the Furlow technique at the age of 10 months. Pre-and post-palatoplasty cleft palate measurements were collected during the procedure. Three blinded speech-language pathologists (SLPs) retrospectively scored the patients from the chart data at age 4. Student t test and receiver operating characteristic curve analysis were used to evaluate the association and predictive capacity between cleft measurements and parameters (M&P) with all VPD criteria.Results:The SLPs found an incidence of VPD according to 7 criteria: hypernasality (11%), audible nasal emission (4%), nasal rustle (14%), compensatory errors (4%), impairment of speech understandability (7%), and impairment of speech acceptability (16%). The SLPs recommended a secondary surgical procedure in 5 patients (9%). A statistically significant association was found between, respectively, 17 and 5 M&P and the occurrence of compensatory errors and audible nasal emission. Our data suggest that the length of the cleft, the cleft area, and the postoperative transversal size of the nasopharynx are the best indicators of the future positivity of VPD criteria.Conclusion:The size of the postoperative transverse nasopharyngeal area during the primary cleft palate procedure may become the focus of the next generation of cleft surgeons to reduce the incidence of VPD.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-26T07:27:54Z
      DOI: 10.1177/1055665620987684
      Issue No: Vol. 58, No. 11 (2021)
       
  • Morphologic Severity of Craniosynostosis: Implications for Speech and
           Neurodevelopment

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      Authors: Damini Tandon, Gary B. Skolnick, Sybill D. Naidoo, Lynn Marty Grames, Mary Michaeleen Cradock, Matthew D. Smyth, Kamlesh B. Patel
      Pages: 1361 - 1369
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1361-1369, November 2021.
      Background:Single-suture craniosynostosis (SSC) can be associated with neurodevelopmental deficits. We examined the correlation between morphologic severity and incidence of speech-language or psychological concerns.Methods:In 62 patients (33 sagittal, 17 metopic, and 12 unicoronal), morphologic severity was determined via preoperative computed tomography (CT). Severity metrics for sagittal, metopic, and unicoronal synostosis were adjusted cephalic index (aCI), interfrontal angle (IFA), and anterior cranial fossa area ratio (ACFR), respectively. Speech-language and psychological concerns were assessed at age ≥4.5 years and defined as recommendation for therapy or monitoring.Results:Mean assessment age was 5.7 years; 32% had a speech-language concern and 44% had a psychological concern; 44% had neither. Sagittal: Mean aCI of those with a speech-language concern (0.62) and those without (0.62) were equivalent (P = .580), as were mean aCI of those with a psychological concern (0.62) and those without (0.62; P = .572). Metopic: Mean IFA with (117.9) and without (125.2) a speech-language concern were equivalent (P = .326), as were mean IFA with (120.2) and without (123.2) a psychological concern (P = .711). Unicoronal: Mean ACFR with (0.65) and without (0.69) a psychological concern (P = .423) were equivalent. However, mean ACFR with (0.74) and without (0.63) a speech-language concern were not (P = .022*). Bivariate rank correlation showed significant association between morphologic severity and speech-language score only for unicoronal synostosis (ρ = .722; P = .008*).Conclusion:A significant portion of patients with SSC had speech-language or psychological concerns. We found no correlation between morphologic severity and incidence of speech-language or psychological concerns for patients with sagittal or metopic synostosis. Morphological severity did correlate with speech concerns in patients with unicoronal synostosis.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-01-20T09:25:35Z
      DOI: 10.1177/1055665620984643
      Issue No: Vol. 58, No. 11 (2021)
       
  • Craniofacial Features in Richieri-Costa-Pereira Syndrome

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      Authors: Milena P. Pardo, Gabriela Lopes dos Santos, Izabel M.M. Carvalho, Kellen Cristine Tjioe
      Pages: 1370 - 1375
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1370-1375, November 2021.
      Objective:Patients with Richieri-Costa-Pereira syndrome (RCPS) present severe craniofacial alterations and frequently require orthodontic and surgical procedures. Thus, this study aims to describe the craniofacial relationships in patients with RCPS.Design:Panoramic radiographs and lateral cephalometric teleradiographs of 7 patients with RCPS and 7 age- and sex-matched nonsyndromic patients were analyzed. Cephalometric measurements were used to determine the size of apical bases, the relationship between them, the pattern of craniofacial growth, and the facial heights of the patients. Interobservers’ concordance was verified by intraclass coefficient. For comparison between the groups, paired t test was employed. P values .05).Conclusion:Patients with RCPS present deficient development of maxilla and mandible when compared with nonsyndromic patients.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-01-28T09:40:54Z
      DOI: 10.1177/1055665620987749
      Issue No: Vol. 58, No. 11 (2021)
       
  • Function of Nasoalveolar Molding Devices in Bilateral Complete Cleft Lip
           and Palate: A 3-Dimensional Maxillary Arch Analysis

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      Authors: Pawinee Tankittiwat, Araya Pisek, Montien Manosudprasit, Pattama Punyavong, Amornrut Manosudprasit, Natthawee Phaoseree, Buddhathida Wangsrimongkol, Poonsak Pisek, Aggasit Manosudprasit
      Pages: 1389 - 1397
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1389-1397, November 2021.
      Objective:This study aimed to observe and analyze the effects of nasoalveolar molding (NAM) on maxillary arch dimensions in patients with bilateral complete cleft lip and palate (BCLP) using 3-dimensional analyses.Design:Retrospective case series.Materials and Methods:Seventeen infants were treated using modified Khon Kaen University presurgical nasoalveolar molding devices (KKU-NAM). Dental casts were evaluated 3 dimensionally at 3 time points: pretreatment (T1), after using modified KKU-NAM for 2 weeks (T2), and before cheiloplasty (T3). Repeated-measures analysis of variance and Friedman test were used to compare the maxillary arch dimensions between time points.Results:Both sides of the cleft width, premaxilla deviation, and premaxilla protrusion had significantly reduced with the use of KKU-NAM. Premaxillary rotation had significantly improved, whereas the arch depth did not change significantly. Premaxilla width, posterior arch width, alveolar length, and height had significantly increased. The anterior arch width, intercanine width, and lateral sulcus width showed no significant changes. The intraclass correlation coefficient used to test the measurements indicated substantial reliability.Conclusion:The modified KKU-NAM is an effective device for reducing the severity of bilateral cleft deformities, especially in the premaxilla area.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-04T09:21:22Z
      DOI: 10.1177/1055665621990184
      Issue No: Vol. 58, No. 11 (2021)
       
  • Australian Orthodontists Utilization and Attitude Toward a Government
           Funded Cleft Scheme

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      Authors: Morgan Wishney, Aziz Sahu-Khan, Peter Petocz, M. Ali Darendeliler, Alexandra K. Papadopoulou
      Pages: 1398 - 1404
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1398-1404, November 2021.
      Objectives:To (1) survey Australian orthodontists about their involvement with a government-funded scheme for patients with clefts, the Medicare Cleft Lip and Cleft Palate Scheme (MCLCPS) and (2) investigate their attitude toward treating patients with clefts and their training in this respect.Design:A 13-question online survey was distributed to members of the Australian Society of Orthodontists. The survey gathered information regarding respondent demographics, the number of MCLCPS-eligible patients seen in the past 12 months and usual billing practices.Results:A total of 96 complete responses were obtained. About 70% of respondents had treated MCLCPS-eligible patients in the past 12 months and 55% saw between 2 and 5 patients during this time. The likelihood of treating patients with clefts increased by a factor of 4.8 (95% CI: 1.2-18.9) if practicing outside of a capital city and 1.5 times for each decade increase in orthodontist’s age (95% CI: 1.0-2.2). The MCLCPS was utilized by 81% of orthodontists with 26% of these respondents accepting rebate only. Most orthodontists felt their university training could have better prepared them to treat patients with clefts. A minority of orthodontists felt that a rebate increase would make them more likely to treat these patients.Conclusions:Australian orthodontists who treat patients with clefts tend to be older and work outside of capital cities. The decision to treat these patients tends to not be financially motived. Specialty orthodontic training programs could improve the preparedness of their graduates to treat patients with clefts.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-02T09:19:41Z
      DOI: 10.1177/1055665621989125
      Issue No: Vol. 58, No. 11 (2021)
       
  • Oral Health-Related Quality of Life Among Sudanese Children Treated for
           Cleft Lip and Palate

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      Authors: Mecheala Abbas Ali, Alwaleed Fadul Nasir, Shaza K. Abass
      Pages: 1405 - 1411
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1405-1411, November 2021.
      Objective:This study compared the oral health-related quality of life (OHRQoL) among children with a cleft lip with or without a cleft palate (CL±P) and a group of their peers. The reliability of the Arabic version of the Child Oral Health Impact Profile Questionnaire (COHIP) was also assessed.Design:A cross-sectional study.Settings:Cleft clinic in a private dental college in Omdurman City, Sudan.Patients:In all, 75 children (mean age 11.3 ± 2.5 years) with a history of CL±P and a group of 150 school children without CL±P (mean age 11.4 ± 2.6 years).Main Outcome Measures:Overall and subscale scores on the Arabic version of the COHIP.Results:Test–retest reliability of COHIP in Arabic was high with an interclass correlation coefficient >0.8. Cronbach α value internal consistency was 0.8 for the total scale and between 0.7 and 0.8 for the subscales. The COHIP score was 89.41 ± 19.97 in children with CL±P and 122.82 ± 9.45 for the control group. Children with CL±P had significantly lower scores on the overall and all subscales when compared to children without CL±P (P ≤ .001). Among the children with CL±P, there were no statistically significant differences on the COHIP based on age and/or gender (P ≥ .05).Conclusions:Children with CL±P had a relatively high OHRQoL, which was lower than that of their peers without CL±P in both the overall scale and all subscales. Gender and age differences had no significant impact on the OHRQoL. The COHIP Arabic version showed appropriate reliability.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-01-20T09:30:36Z
      DOI: 10.1177/1055665620987694
      Issue No: Vol. 58, No. 11 (2021)
       
  • Interpersonal Relationship Experiences in Adults Born With Cleft Lip
           and/or Palate: A Whole of Life Survey in the United Kingdom

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      Authors: Kenny Ardouin, Matt Hotton, Nicola Marie Stock
      Pages: 1412 - 1421
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1412-1421, November 2021.
      Objectives:Previous research has suggested that adults with a cleft lip and/or palate (CL/P) may find their familial, friend, collegial, and/or romantic relationships impacted by their condition. Building on this prior work, this study examined the self-reported interpersonal experiences of a national sample of adults born with CL/P.Design:An online survey was designed by the Cleft Lip and Palate Association in collaboration with the Centre for Appearance Research. Data from 181 participants were included in the present study, collected between July and October 2018. Qualitative data were analyzed using inductive content analysis, while quantitative data were analyzed using descriptive statistics and independent t tests.Results:Most participants reported having good relationships with family members and existing friends with similar patterns of marriage as the general population. However, some experienced difficulties initiating new friendships and romantic relationships. Participants described bullying and discrimination in adulthood, most often in public settings and the workplace. Participants reported lower satisfaction with intimate relationships and less secure attachment compared to the general population. Concerns regarding the heritability of CL/P and its impact on the decision to have a family were also reported.Conclusions:Individuals with CL/P are at risk of negative social experiences, which if not appropriately addressed may impact psychological well-being in adulthood. The findings illustrate the importance of routine psychosocial support from childhood onward to help individuals with CL/P build social skills, self-esteem, and social confidence and to develop mutually fulfilling friendships and intimate relationships. Additionally, societal campaigns to raise awareness and target discrimination may be helpful.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-04T09:19:53Z
      DOI: 10.1177/1055665620987109
      Issue No: Vol. 58, No. 11 (2021)
       
  • Seasonal Influence on the Numbers of Gender-Related Orofacial Cleft
           Conceptions in the Netherlands

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      Authors: Lisanne M. van der Lek, Shariselle M.W. Pool, Kim de Jong, Christl Vermeij-Keers, Chantal M. Mouës-Vink
      Pages: 1422 - 1429
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1422-1429, November 2021.
      Background:In the multifactorial etiology of orofacial clefts (OFCs), environmental factors play an important role. To trace the influence of these factors, the timing of the cell biological mechanisms that occur during embryological development of the primary and secondary palates must be taken into account. That is, the fusion process of the facial and palatal processes, respectively, followed by their differentiation into bone and musculature, which take place during the first trimester of pregnancy. During this period, harmful seasonal influences such as viral infections and vitamin deficiencies could induce OFC in the embryo.Aims:The aim of this study is to find out whether a seasonal conception period with an increased risk of OFC development exists, particularly gender related.Methods:This was a retrospective cross-sectional study on children with OFC born in the Netherlands from 2006 to 2016. Total conception rates of live births in the Netherlands were used as a control group. χ2 tests were performed to analyze monthly and seasonal differences. Males and females, positive and negative family history and subphenotype groups based on fusion and/or differentiation (F- and/or D-) defects, and their timing in embryogenesis were analyzed separately.Results:In total, 1653 children with OFC, 1041 males and 612 females, were analyzed. Only males with FD-defects showed a significant seasonal variation with an increase in conceptions during spring, most often in May.Conclusions:Males with FD-defects showed a significant seasonal variation with an increase in conceptions during spring. No other seasonal trends could be demonstrated.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-01-20T09:29:30Z
      DOI: 10.1177/1055665620987693
      Issue No: Vol. 58, No. 11 (2021)
       
  • Registry-Based Study of Prevalence of Cleft Lip/Palate in Thailand from
           2012 to 2015

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      Authors: Pornpoj Fuangtharnthip, Wannapong Chonnapasatid, Sasipa Thiradilok, Somchai Manopatanakul, Somchit Jaruratanasirikul
      Pages: 1430 - 1437
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1430-1437, November 2021.
      Objective:Two main objectives were established. First objective was to determine the prevalence of the cleft lip and/or cleft palate (CL/P) in Thailand from 2012 to 2015 using the orofacial clefts (OFCs) registry and civil registration. Second objective was to conduct a quality control of this OFC registry especially for the Birth Defects Registration (BDR).Design:Registry-based survey.Setting:Analyzing data from the Thailand National Health Security Office.Participants:Registered patients with CL/P in Thailand from 2012 to 2015.Intervention:NoneMain Outcome Measure:Duplicated records were verified using National Identity Number (Thai ID#) and date of birth. The prevalence of CL/P and specific phenotypes was then calculated. From this prevalence estimate method, quality assurance of the OFCs registry was possible.Results:For the main outcome, the population-weighted pool prevalence of CL/P was 2.14 per 1000 live births (95% confidence interval of 2.08-2.20). Thai ID# and expense reimbursement systems were the main factors driving this cases capturing. However, this OFCs registration still requires active case finding with clinical verification, improvement of staff training and databases networking.Conclusions:This study reported a very high CL/P prevalence of Thailand. Strengths and limitations of these OFCs registry and BDR were described.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-01-25T09:34:26Z
      DOI: 10.1177/1055665620987677
      Issue No: Vol. 58, No. 11 (2021)
       
  • Treacher Collins Syndrome in the United States: Examining Incidence and
           Inpatient Interventions

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      Authors: Lauren Reid, William Carroll
      Pages: 1438 - 1442
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1438-1442, November 2021.
      Objective:Advancements have been made in the care of patients with Treacher Collins syndrome (TCS), but epidemiological data are lacking given its rarity. A national database provides a valuable opportunity for studying the incidence of rare craniofacial conditions. We sought to evaluate disease incidence of phenotypically severe cases and the frequency of the most common associated diagnoses and interventions.Design:The 2016 Kids’ Inpatient Database (KID), Healthcare Cost and Utilization Project, Agency for Healthcare Research and Quality was the first version to include updated International Classification of Diseases-Tenth Edition (ICD-10) coding encompassing a unique code for TCS. The 2016 KID was queried for the unique code. Incidence was calculated using national estimates. Diagnosis and procedure codes were pooled and analyzed.Patients:A total of 266 discharge cases with a diagnosis code for TCS (Q75.4).Main Outcomes:Disease incidence and the most frequent diagnosis and procedure codes.Results:The estimated incidence of children born with a diagnosis of TCS was approximately 1 in 80,000. Two of the three most common ICD-10 diagnosis codes were tracheostomy status and obstructive sleep apnea. The most common procedures performed were airway examination procedures.Conclusions:Treacher Collins syndrome is a rare craniofacial disorder requiring complex multidisciplinary management. Phenotypically severe cases requiring inpatient management may be rarer than previous estimates suggest. Inpatient airway interventions are very common, and multidisciplinary team members should anticipate the degree of potential airway risk these patients pose.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-31T08:59:16Z
      DOI: 10.1177/1055665621998138
      Issue No: Vol. 58, No. 11 (2021)
       
  • Custom-Made Nasal Retainer Using Latex Nelaton Urine Catheter After the
           Cheiloplasty

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      Authors: Joon Seok Oh, Jeehyeok Chung, Jeong Hyun Ha, Hyo Kyung Yoo, Sukwha Kim
      Pages: 1443 - 1445
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1443-1445, November 2021.
      Nasal retainers are common tools used in managing patients with cleft lip. The significance of nasal retainer in preventing nostril collapse or stenosis to maintain a symmetrical nose after the surgical procedures is already well known. We came up with a way to create a nasal retainer using a latex nelaton catheter. Custom-made nasal retainer using latex nelaton catheter was used postoperatively on a 10-month-old infant with median cleft lip after cheiloplasty. In postoperative day 7, her nostrils were large enough for premade silicone nasal retainer to fit. She was discharged with instructions given to use the retainer for 6 months. Custom-made nasal retainer can be used as an alternative to premade nasal retainers for patients with wide columella or small nostril cavities, or who cannot afford premade retainers.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-12T10:22:35Z
      DOI: 10.1177/1055665621992673
      Issue No: Vol. 58, No. 11 (2021)
       
  • Congenital Midline Cervical Cleft: A Variant of Tessier Number 30 Cleft
           Causing Micrognathia

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      Authors: Cristiano Tonello, Ines Correia Pinto de Matos, Leonardo Bezerra Feitosa, Adriano Porto Peixoto, Nivaldo Alonso
      Pages: 1446 - 1451
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1446-1451, November 2021.
      Congenital midline cervical cleft is a rare and generally isolated malformation of the ventral neck region with no clear etiology established. Mandibular deformities, such as micrognathia, could be considered as a consequence of a cleft cervical contracture. Complete surgical excision of the subcutaneous fibrous cord at an early age is the primary treatment modality, minimizing growth development problems on surrounding affected tissue. The aim of this study is to describe the clinical, surgical, and histological findings in a female child with congenital midline cervical cleft along with a relevant literature review. Three years follow-up after surgery exhibited satisfactory functional and cosmetic results.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-01-13T01:10:07Z
      DOI: 10.1177/1055665620987412
      Issue No: Vol. 58, No. 11 (2021)
       
  • Dens Invaginatus in Patients With Cleft Lip and Palate: A Case Series

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      Authors: Ilana M. Ickow, Steven Zinn, J. Matthew Stacy, Brian Martin, Joseph E. Losee, Alene D’Alesio, Jane Soxman, Lindsay A. Schuster
      Pages: 1452 - 1458
      Abstract: The Cleft Palate-Craniofacial Journal, Volume 58, Issue 11, Page 1452-1458, November 2021.
      Dens invaginatus is a rare developmental anomaly characterized by an infolding of the enamel organ within the crown or root of a tooth, and it is an example of a dental anomaly that has a higher incidence in patients with CL/P. If undiagnosed, dens invaginatus can lead to severe, acute pain and pulpal necrosis since it can permit direct entry of bacteria into the dental pulp. Treatment of dens invaginatus includes prophylactic sealant or composite restoration, endodontic therapy if pulpal involvement has already occurred, or extraction if aberrant tooth morphology precludes endodontic therapy. Few studies report on the incidence of dens invaginatus in patients with CL/P. The purpose of this article is to describe 4 cases of dens invaginatus in patients with CL/P which were encountered in a cleft-craniofacial orthodontic clinic. Each case describes dens invaginatus in a maxillary lateral incisor, and treatments ranged from sealant application to endodontic therapy to extraction. These cases highlight the importance of awareness of this dental anomaly among cleft team providers to facilitate early diagnosis in patients with CL/P.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-05T08:56:50Z
      DOI: 10.1177/1055665621998534
      Issue No: Vol. 58, No. 11 (2021)
       
  • Predictors of Complication Following Cleft Lip and Palate Surgery in a
           Low-Resource Setting: A Prospective Outcomes Study in Nicaragua

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      Authors: Marco A. Swanson, Allyn Auslander, Tatiana Morales, Breanna Jedrzejewski, William P. Magee, Armando Siu, Ruben Ayala, Jordan W. Swanson
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundHigher rates of postoperative complication following cleft lip or palate repair have been documented in low resource settings, but their causes remain unclear. This study sought to delineate patient, surgeon, and care environment factors in cleft complications in a low-income country.DesignProspective outcomes study.SettingComprehensive Cleft Care Center.PatientsCandidate patients presenting for cleft lip or palate repair or revision.InterventionsPatient anthropometric, nutritional, environmental and peri- and post-operative care factors were collected. Post-operative evaluation occurred at standard 1-week and 2-month postoperative intervals.Main Outcome MeasuresComplication was defined as fistula, dehiscence and/or infection.ResultsAmong 408 patients enrolled, 380 (93%) underwent surgery, of which 208 (55%) underwent lip repair (124) or revision (84), and 178 (47%) underwent palate repair (96) or revision (82). 322 (85%) were evaluated 1 week and 166 (44%) 2 months postoperatively. 50(16%) complications were identified, including: 25(8%) fistulas, 24(7%) dehiscences, 17(5%) infections. Mid-upper arm circumference (MUAC) ≤12.5 cm was associated with dehiscence after primary lip repair (OR = 28, p = 0.02). Leukocytosis ≥11,500 on pre-operative evaluation was associated with dehiscence (OR = 2.51, p = 0.04) or palate revision fistula (OR = 64, p 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-18T12:13:57Z
      DOI: 10.1177/10556656211046810
       
  • Treatment Outcome in Bilateral Cleft lip and Palate Patients Evaluated
           With the Huddart-Bodenham Scoring System and the Bilateral Cleft lip and
           Palate Yardstick: A Systematic Review

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      Authors: Wenying Kuang, Miranda Aarts, Anne Marie Kuijpers-Jagtman, Hong He, Edwin M. Ongkosuwito
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivesTo assess treatment outcome (transversal and sagittal dental arch relationships) and its determinants in complete bilateral cleft lip and palate (BCLP) evaluated with the modified Huddart-Bodenham scoring system and the BCLP Yardstick.Materials and methodsMultiple electronic databases were searched without time limitation. Randomized clinical trials, cohort and case control studies using BCLP Yardstick and/or modified Huddart-Bodenham system to judge treatment outcome of patients with BCLP were included. The Risk of Bias in Nonrandomized Studies of Interventions tool and Grading of Recommendations, Assessment, Development, and Evaluation was used.ResultsOf the 528 studies identified by the electronic search, only eight retrospective studies met the inclusion criteria and were included. A total of 12 cleft centers were represented. All treatment protocols differed and background information was underreported. The results for the BCLP yardstick showed that all except the centers in New Zealand had a mean score lower than 3, indicating good treatment results. However, these studies had a moderate to high risk of bias. The modified Huddart-Bodenham scores were negative in all studies. No further meta-analysis was done due to heterogeneity and high risk of bias. The quality of evidence was graded as very low.ConclusionResults for the dental arch relationship of studies in complete BCLP and possible determinants were not synthesized due to very low quality of evidence. Clinical research for patients with BCLP should focus on sound methodological designs to enable evidence-based decision making to improve treatment for patients with BCLP and thereby hopefully their quality of life.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-18T12:13:40Z
      DOI: 10.1177/10556656211041883
       
  • Race-Based Differences in the Utilization and Timing of Secondary Cleft
           Procedures in the United States

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      Authors: Connor J. Peck, Navid Pourtaheri, Yassmin Parsaei, Arvind U. Gowda, Jenny Yang, Joseph Lopez, Derek M. Steinbacher
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective: Secondary procedures optimize outcomes following cleft lip and palate (CL/P) repair. We analyzed the association between race and the utilization and timing of revisionary/secondary cleft surgeries in the United States.DesignPrimary CL/P repair, revisions, and secondary procedures—cleft rhinoplasty, speech surgery, and alveolar bone grafting (ABG)—performed from 2014–2018 were identified from the Pediatric National Surgical Quality Improvement Program (NSQIP) database. Utilization estimates were derived via univariable and multivariable logistic regression. A Kruskal-Wallis rank-sum test and multivariable linear regression were used to assess differences in timing for each procedure cohort.Main Outcome MeasuresThe primary outcome measures were the odds of a patient being a certain race/ethnicity, and the age at which patients of different race/ethnicity receive surgery.ResultsThere were 23 780 procedures analyzed. After controlling for sex, diagnosis, and functional status, there were significant differences in utilization estimates across procedure groups. Primarily, utilization was lowest in patient who were Black for cleft rhinoplasty (OR = 0.70, P = .023), ABG (OR = 0.44, P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-18T04:47:50Z
      DOI: 10.1177/10556656211047134
       
  • Long-term Morphological Changes of the Velum and the Nasopharynx in
           Patients With Cleft Palate

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      Authors: Takeshi Harada, Tadashi Yamanishi, Takayuki Kurimoto, Setsuko Uematsu, Yuri Yamamoto, Naoko Inoue, Juntaro Nishio
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo investigate long-term morphological changes in the soft palate length and nasopharynx in patients with cleft palate. We hypothesized that there would be differences in the morphological development of the soft palate and nasopharynx between patients with and without cleft palate and that these developmental changes would negatively affect the soft palate length to pharyngeal depth ratio involved in velopharyngeal closure for patients with cleft palate.DesignRetrospective, case-control study.SettingInstitutional practice.PatientsNinety-two patients (Group F) with unilateral cleft lip, alveolus, and palate and 67 patients (Group CLA) with unilateral cleft lip and alveolus not requiring palatoplasty were included.Main Outcome MeasuresThe soft palate length, nasopharyngeal size, and soft palate length to pharyngeal depth ratio were measured via lateral cephalograms obtained at three different periods.ResultsGroup F showed a shorter soft palate length and smaller nasopharyngeal size than Group CLA at all periods. Both these parameters increased with age, but the increase in amount was significantly less in Group F compared with that in Group CLA. The soft palate length to pharyngeal depth ratio in Group F decreased with age.ConclusionsIn patients with cleft palate, the soft palate length to pharyngeal depth ratio, which is involved in velopharyngeal closure, can change with age. Less soft palate length growth and unfavorable relationship between the soft palate and nasopharynx may be masked in early childhood but can manifest later on with age.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-18T04:47:19Z
      DOI: 10.1177/10556656211045287
       
  • Behavioral and Social Functioning of Children With and Without Positional
           Plagiocephaly: Late Infancy to School Age

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      Authors: Cindy Ola, Matthew L. Speltz, Brent R. Collett
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivePositional plagiocephaly and/or brachycephaly (PPB) is associated with cognition, motor, and other developmental outcomes, but little is known about the social-behavioral adjustment of children with PPB. The primary aim of this study was to compare the social-behavioral development of preschool and school-age children with and without PPB and to examine the potential moderating effects of PPB severity on group differences.DesignTwo hundred twenty children with a history of PPB and 164 controls participated in at least one behavioral assessment at 4-11 months, 18 months, 36 months, and 7 years. The frequencies of observed problem behaviors and social competence were estimated using the Child Behavior Checklist Ages 1.5-5 (CBCL/1.5-5), Caregiver-Teacher Report Form(C-TRF), CBCL/6-18, and Teacher Report Form.ResultsChildren with PPB were similar to controls on the internalizing, externalizing, or total problems composites. At 7 years, CBCL/6-18 total competence scores were significantly lower in children with histories of PPB than controls. In analyses stratified by PPB severity, we found that children with moderate/severe PPB had slightly higher scores on the C-TRF internalizing scale at 36 months and lower total competence scores at age 7 years. Children who had a history of mild PPB were similar to controls on all outcomes.ConclusionsThis study is the first to examine social and behavioral outcomes in a large cohort of children with and without a history of PPB. We found limited evidence of an association between PPB and parent and teacher-reported social-emotional and behavioral adjustment through early school-age.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-14T10:07:25Z
      DOI: 10.1177/10556656211043739
       
  • Double-opposing Z-Plasty Extended with a Pedicled Buccal Fat Pad Flap for
           Correcting Velopharyngeal Insufficiency after Primary Palatoplasty

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      Authors: Hojin Park, Jin Mi Choi, Tae Suk Oh
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionFurlow double-opposing Z-plasty (DOZ) lengthens the soft palate; however, this lengthening is achieved at the expense of increased mucosal flap tension. Thus, its use is limited in patients with severe tension applied on mucosal flap after DOZ. In this study, DOZ was combined with a buccal fat pad (BFP) flap to maximize palatal lengthening and muscle repositioning.MethodsThis study included patients who underwent surgical correction for velopharyngeal insufficiency between December 2016 and February 2019. Patients with more than moderate degree hypernasality following primary palatoplasty were included in the study. Patients younger than 4 years of age, those with a submucous cleft palate, or syndromic patients were excluded. Speech outcomes were investigated for those who underwent DOZ only (DOZ group, n = 17) and those in whom a BFP was used (BFP group, n = 15) pre- and postoperatively. The velopharyngeal gaps between the uvula and pharyngeal wall were measured before and immediately after surgery to estimate the palatal length.ResultsMost patients who received a BFP showed improvement in hypernasality. However, the hypernasality of the DOZ group was more severe than that of the BFP group (p = 0.023). The extent of palatal lengthening was 4.4 ± 1.7 mm and 7.5 ± 2.1 mm in the DOZ and BFP groups, respectively (p = 0.001).ConclusionsBFPs reduced the tension of the DOZ mucosal flap and maximized palatal lengthening and muscle repositioning. They promoted velopharyngeal closure in patients with moderate and moderate-to-severe velopharyngeal insufficiency. Hence, our method improves the surgical outcomes of patients with velopharyngeal insufficiency after primary palatoplasty.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-12T12:41:13Z
      DOI: 10.1177/10556656211047139
       
  • Emergency Response Protocols for Overseas Outreach Settings: Global Smile
           Foundation Strategy

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      Authors: Elsa M. Chahine, Nikhil D. Shah, Omar S. Al Abyad, Raj M. Vyas, Usama S. Hamdan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Non-governmental organizations (NGOs) aim to alleviate unmet global disease burden and promote collaboration between visiting and host countries. Well-executed emergency response protocols are foundational to providing safe and quality care in an unpredictable global setting. Global Smile Foundation (GSF) instituted a protocol in 2012 based on over three decades of cleft care experience. Here, we update this protocol and provide information on how to address special circumstances such as humanitarian crises and disease outbreaks. The GSF response protocol was developed in conjunction with surgeons, anesthesiologists, nurses, and administrators to ensure all team members are adequately prepared to respond to emergency situations in global outreach. This protocol provides information on pre-departure preparation, onsite setup, operative precautions, and post-departure debriefings. Emphasis is placed on a standardized, reproducible workflow that accounts for necessary site-specific adjustments. Strategies include emergency simulations, site-specific safety checklists, standardized operating room protocols, and well-defined individual responsibilities. The authors also provide anticipatory guidance in addressing unexpected circumstances, such as disease outbreak and natural disaster, during global outreach. In conclusion, a proactive and systematic approach to emergency response and prevention is vital to minimizing morbidity and mortality during surgical outreach initiatives. The GSF protocol represents a reproducible approach that other organizations can adopt and adapt to their unique needs.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-12T03:04:18Z
      DOI: 10.1177/10556656211049998
       
  • Quality of Life and Temporomandibular Disorders in Patients With Skeletal
           Class III Malocclusion With Cleft Lip and Palate

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      Authors: Bernardo Olsson, Isabela Polesi Bergamaschi, Erika Calvano Küchler, Aline Monise Sebastiani, Guilherme dos Santos Trento, Delson Joao da Costa, Nelson Luis Barbosa Rebellato, Rafaela Scariot
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe aim of the study was to assess the quality of life (QOL), oral health-related QOL (OHRQOL), temporomandibular disorders (TMDs), and psychological factors in patients with skeletal Class III malocclusion with cleft lip and palate (CLP) and without CLP.DesignCase–control.SettingPrimary care, institutional practice.PatientsOne hundred thirty-six patients with skeletal Class III malocclusion with CLP (n = 68) and without CLP (n = 68).Main outcome measuresQOL and OHRQOL were assessed using the World Health Organization Quality of Life-BREF (WHOQOL-BREF) questionnaire and the Oral Health Impact Profile-14 questionnaire, respectively. TMDs and psychological factors were assessed using the Research Diagnostic Criteria for TMD (RDC/TMD).ResultsNo differences in QOL were found between the groups (P > 0.05). Patients with CLP reported a better OHRQOL (P = 0.025) in the physical pain, physical disability, and psychological disability domains (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-12T03:04:05Z
      DOI: 10.1177/10556656211043429
       
  • Cost of Cleft Team Care at an Academic Children’s Hospital

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      Authors: Jennifer Lee, Gary B. Skolnick, Sybill D. Naidoo, Sibyl Scheve, Cheryl Grellner, Alison Snyder-Warwick, Kamlesh B. Patel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundThe financial burden of cleft-craniofacial team care is substantial, and high costs can hinder successful completion of team care.SolutionCollaboration with multiple stakeholders including providers, insurers, and patient guardians, as well as hospital administrators, is critical to increase patient retention and improve final clinical outcomes.What We Do That is NewAt our cleft and craniofacial center, charges for a team care visit fall into one of three categories—hospital fees, professional fees, or external fees. There are four types of hospital fees depending on (1) whether the patient is new or returning, and (2) whether the patient saw ≤4 or ≥5 providers. To further elucidate the financial burden (out-of-pocket costs) directly borne by families of children with cleft lip and/or palate, we conducted a retrospective review of billing records of team care visits made between September 2019 and March 2020. Out-of-pocket costs for a single team care visit (on a commercial insurance plan) ranged from $4 to approximately $1220 and had a median (IQR) of $445 ($118, $749).
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-07T12:01:58Z
      DOI: 10.1177/10556656211046815
       
  • Heritability Analysis in Twins Indicates a Genetic Basis for
           Velopharyngeal Morphology

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      Authors: Myoung Keun Lee, Chenxing Liu, Elizabeth J. Leslie, John R. Shaffer, Jamie L. Perry, Seth M. Weinberg
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe velopharyngeal mechanism is comprised of several muscular components that act in a coordinated manner to control airflow through the nose and mouth. Proper velopharyngeal function is essential for normal speech, swallowing, and breathing. The genetic basis of normal-range velopharyngeal morphology is poorly understood. The purpose of this study was to estimate the heritability of velopharyngeal dimensions.MethodWe measured five velopharyngeal variables (velar length, velar thickness, effective velar length, levator muscle length and pharyngeal depth) from MRIs of 155 monozygotic and 208 dizygotic twin pairs and then calculated heritability for these traits using a structural equation modeling approach.ResultsThe heritability estimates were statistically significant (95% confidence intervals excluded zero) and ranged from 0.19 to 0.46. There was also evidence of significant genetic correlations between pairs of traits, pointing to the influence of common genetic effects.ConclusionsThese results indicate that genetic factors influence variation in clinically relevant velopharyngeal structures.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-10-04T11:02:23Z
      DOI: 10.1177/10556656211045530
       
  • Improving Psychosocial Risk Assessment and Service Provision for
           Craniofacial Team Patients: A Quality Improvement Project

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      Authors: Canice E. Crerand, Meghan O’Brien, Hillary M. Kapa, Ari N. Rabkin, Amanda Smith, Richard E. Kirschner, Gregory D. Pearson, Jahnavi Valleru, Adriane L. Baylis
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo improve psychosocial risk assessment and service provision for children with craniofacial conditions presenting for annual interdisciplinary team visits.DesignInstitute for Healthcare quality improvement model.SettingU.S. pediatric academic medical center.ParticipantsCaregivers of children ages 0-17 years with craniofacial conditions presenting for 1692 team visits between August 2017 and July 2019.InterventionsKey drivers included: (1) standardizing pre-visit triage processes; (2) administering the Psychosocial Assessment Tool-Craniofacial Version (PAT-CV); (3) utilizing PAT-CV scores in real time to add patients to psychosocial provider schedules; and (4) family education. Interventions included improving patient screening, increasing PAT-CV completion rate, altering clinic flow, providing patient and parent education about psychosocial services, and altering team member roles to fully integrate PAT-CV administration and scoring in the clinic.Main Outcome MeasuresThe primary outcome was the percentage of patients identified for psychosocial consultations via nurse triage, PAT-CV score, family or provider request who completed consultations. The secondary outcome was the percentage of patients completing needed psychosocial consultations based on elevated PAT-CV scores.ResultsUse of the PAT-CV resulted in an increase in the percentage of patients with elevated psychosocial risk who received a psychosocial consultation from 86.7% to 93.4%. The percentage of children receiving psychosocial consultation at their annual team visit due to elevated PAT-CV scores increased from 72% to 90%.ConclusionsIntegrating a validated psychosocial risk screening instrument can improve risk identification and psychosocial consultation completion. A combination of risk screening approaches may be indicated to identify patients in need of psychosocial services.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-30T10:55:22Z
      DOI: 10.1177/10556656211043006
       
  • Utilization of 3D MRI for the Evaluation of Sphincter Pharyngoplasty
           Insertion Site in Patients With Velopharyngeal Dysfunction

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      Authors: Kazlin N. Mason, John E. Riski, Joseph K. Williams, Richard A. Jones, Jamie L. Perry
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveSphincter pharyngoplasty is a surgical method to treat velopharyngeal dysfunction. However, surgical failure is often noted and postoperative assessment frequently reveals low-set pharyngoplasties. Past studies have not quantified pharyngoplasty tissue changes that occur postoperatively and gaps remain related to the patient-specific variables that influence postoperative change. The purpose of this study was to utilize advanced three-dimensional imaging and volumetric magnetic resonance imaging (MRI) data to visualize and quantify pharyngoplasty insertion site and postsurgical tissue changes over time.DesignA prospective, repeated measures design was used for the assessment of craniometric and velopharyngeal variables postsurgically. Imaging was completed across two postoperative time points. Tissue migration, pharyngoplasty dimensions, and predictors of change were analyzed across imaging time points.ResultsSignificant differences were present between the initial location of pharyngoplasty tissue and the pharyngoplasty location 2 to 4 months postoperatively. The average postoperative inferior movement of pharyngoplasty tissue was 6.82 mm, although notable variability was present across participants. The pharyngoplasty volume decreased by 30%, on average.ConclusionsInferior migration of the pharyngoplasty tissue was present in all patients. Gravity, scar contracture, and patient-specific variables likely interact, impacting final postoperative pharyngoplasty location. The use of advanced imaging modalities, such as 3D MRI, allows for the quantification and visualization of tissue change. There is a need for continued identification of patient-specific factors that may impact the amount of inferior tissue migration and scar contracture postoperatively.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-27T10:22:40Z
      DOI: 10.1177/10556656211044656
       
  • Maternal Cigarette Smoking and Cleft Lip and Palate: A Systematic Review
           and Meta-Analysis

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      Authors: Matthew Fell, Kyle Dack, Shaheel Chummun, Jonathan Sandy, Yvonne Wren, Sarah Lewis
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivesA systematic review and meta-analysis to determine the association between active maternal smoking and cleft lip and palate etiology.Data sourcesMedline, Embase, Web of Science and the Cochrane Library from inception to November, 2020.Study selectionObservational studies of cigarette smoking habits in pregnant women. Outcomes included cleft lip and/or palate, cleft lip  ±  palate and cleft palate only.Data analysisPublication bias analyses were performed and the Newcastle Ottawa scales were used to assess study quality. Fixed or random effect models were used in the meta-analysis, dependent on risk of statistical heterogeneity.ResultsForty-five studies were eligible for inclusion of which 11 were cohort and 34 were case–control studies. Sixteen studies were of sufficient standard for inclusion in the meta-analysis. The summary odds ratio for the association between smoking and cleft lip and/or palate was 1.42 (95%CI 1.27-1.59) with a population attributable fraction of 4% (95%CI 3%-5%). There was limited evidence to show a dose–response effect of smoking.ConclusionsThis review reports a moderate association between maternal smoking and orofacial cleft but the overall quality of the conventional observational studies included was poor. There is a need for high quality and novel research strategies to further define the role of smoking in the etiology of cleft lip and palate.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-27T01:08:31Z
      DOI: 10.1177/10556656211040015
       
  • Alveolar Cleft Reconstruction Using Double Iliac Corticocancellous Bone
           Plates Grafting Technique in Mixed Dentition Phase

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      Authors: Mohammed Omara, Mamdouh Ahmed, Mohamed Shawky, Sherif Ali
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe primary goal of maxillary alveolar cleft reconstruction in patients with cleft lip/palate at the phase of mixed dentition is to build bone in the cleft area which in turn allows closure of the oronasal fistula, establishes arch continuity, and improve maxillary stability. This study aimed to evaluate the double iliac corticocancellous bone plates grafting technique for initial alveolar cleft grafting.DesignThis prospective study was conducted on 12 consecutive patients with unilateral complete alveolar cleft and previous cleft lip and palate corrective surgery.InterventionFor all patients, the iliac crest graft was harvested and cut into 2 cortical bone plates and adapted labially and palatally. Both plates were fixed with screws then the gap between the 2 plates was filled with cancellous bone. The grafted side was compared to the contralateral side 9 months postoperatively regarding labio-palatal alveolar width and bone density, while the 9 months postoperative graft height was compared to the immediate postoperative height using computed tomography scans.ResultsAll grafted sides showed similarity to the contralateral sides regarding both alveolar width and bone density with minimal crestal bone resorption.ConclusionThe double iliac corticocancellous bone plate grafting technique seems to be a reliable method for alveolar cleft reconstruction with adequate bone quality and contour.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-24T10:58:44Z
      DOI: 10.1177/10556656211042160
       
  • Validation of Skully Care as a Fast Method for Quantifying Positional
           Cranial Deformities

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      Authors: Léon N.A. Van Adrichem, Sophia A.J. Kronig, Otto D.M. Kronig
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveCraniofacial measuring is valuable for diagnosis and evaluation of growth and treatment of positional skull deformities. Plagiocephalometry (PCM) quantifies skull deformities and is proven to be reliable and valid. However, PCM needs direct skin contact with thermoplastic material, is laborious and time-consuming. Therefore, Skully Care (SC) was developed to measure positional skull deformities with a smartphone application.DesignSC is retrospectively compared to PCM.SettingPediatric physiotherapy centers.PatientsAge ≤1 year, analyzed or treated for positional skull deformities.InterventionsA total of 60 skull shape analyses were performed.Main Outcome MeasuresThe main outcome measures employed are Pearson correlation coefficient between cranial vault asymmetry index (CVAI; in SC) and oblique diameter difference index (ODDI; in PCM) and between cranial index (CI; in SC) and cranial proportional index (CPI; in PCM). Mann–Whitney U test determined difference of time consumption between PCM and SC.ResultsHigh correlation was found between CVAI and ODDI (r = 0.849; P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-24T02:55:18Z
      DOI: 10.1177/10556656211035022
       
  • Evaluating Psychosocial Problems in School-Age Children with Cleft Lip and
           Palate in Bandung, Indonesia Using CBCL/6-18

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      Authors: Hardisiswo Soedjana, Kristaninta Bangun, Sitha Christine
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundCleft lip with or without palate (CL  ±  P) may impact children’s eating, drinking, speaking, breathing, and hearing. We aim to evaluate psychosocial problems in Indonesian cleft center school-age patients identified after one or more surgical interventions.MethodsThis is a cross-sectional study of parent report of patients with unilateral CL  ±  P who had cleft surgery from 2011 to 2016 in the Bandung Cleft Center using the Bahasa Indonesia version of CBCL/6-18 questionnaire. Descriptive statistics were completed based on measure norms and score ranges.ResultsThere were 104 participants (56.7% male) with a median age of 8 years old and 73.0% had unilateral cleft of lip, gum, and palate. We found that speech and appearance problems were not perceived by parents for 34.6% of participants after undergoing surgery. The majority of parents reported normal range scores for the Social scale (93.3%) and the School scale (92.3%). In contrast, largely due to the restrictions in the covid-19 pandemic, 78.8% of the patients had below normal range scores for the Activities scale. Borderline or Clinical range scores were reported for 6.7% of children on the Problem Items section and 15.4% of parents endorsed one or more Critical Items about their children, which indicate significant behavioral concerns.ConclusionIn this study, we found 6.7% of the school-age children population with CL/P had psychosocial problems. The result of this study hopefully can shed some light in the long-term psychosocial conditions of the CL/P children post-operatively.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-22T11:25:45Z
      DOI: 10.1177/10556656211040703
       
  • Clinical Practice Trends and Postoperative Outcomes in Primary Cleft
           Rhinoplasty

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      Authors: Hossein E. Jazayeri, Joseph Lopez, Navid Pourtaheri, Kevin C. Lee, Connor J. Peck, David L. Best, Jason W. Yu, Arun K. Gosain, Zachary S. Peacock, Sean P. Edwards, Derek M. Steinbacher
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      PurposeOptimal correction of the cleft nasal deformity remains challenging. The purpose of this study was to examine the practice patterns and postoperative course of patients undergoing cleft lip repair with rhinoplasty compared to those who have primary lip repair without rhinoplasty.Methods and MaterialsA retrospective cohort study was conducted based on the Kids’ Inpatient Database. Data were collected from January 2000 to December 2011 and included infants aged 12 months and younger who underwent cleft lip repair. The predictor variable was the addition of rhinoplasty at primary cleft lip repair. Primary outcome variables included hospital setting, year, and admission cost, while secondary outcome variables included length of stay and postoperative complication rate. Independent t-tests and chi-squared tests were performed. Continuous variables were analyzed by multiple linear regression models.ResultsThe study sample included 4559 infants with 1422 (31.2%) who underwent primary cleft rhinoplasty. Over time, there was a significant increase in the proportion of cleft lip repairs accompanied by a rhinoplasty (p 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-22T11:23:59Z
      DOI: 10.1177/10556656211032196
       
  • Dynamic Change in Oral Microbiota of Children With Cleft Lip and Palate
           After Alveolar Bone Grafting

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      Authors: Kejia Zhang, Xuan Zhou, Jinwei Qin, Weibing Zhang, Yongchu Pan, Hua Wang, Jiuxiang Lin, Luwei Liu, Yilin Jia
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo investigate the longitudinal influence of alveolar bone grafting on the oral microbiota of children with cleft lip and palate (CLP).Materials and methodsTwenty-eight children with nonsyndromic CLP were recruited and underwent secondary alveolar bone grafting at the first time. Unstimulated saliva and plaque samples were collected from the subjects preoperatively and at 2 days, 1 month, and 3 months postoperatively. The v3–v4 hypervariable regions of the 16S rRNA gene from bacterial DNA were sequenced using the Illumina MiSeq sequencing platform.ResultsThe alpha diversity of the saliva and plaque microbiota was significantly decreased at 2 days postoperatively and then increased at 1 and 3 months postoperatively. The saliva and plaque microbiota compositions at 2 days postoperatively differed from those at the other time points, and the microbiota compositions at 1 and 3 months postoperatively showed a gradual shift toward the preoperative composition. The saliva, but not plaque, microbiota composition 3 months postoperatively was similar to that preoperatively.ConclusionThe effect of secondary alveolar bone grafting on the plaque microbiota in children with CLP lasted longer than the saliva microbiota. Alveolar bone grafting altered the saliva microbiota in children with CLP within 3 months postoperatively.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-15T05:32:51Z
      DOI: 10.1177/10556656211044396
       
  • Orthognathic Surgery: My Experience of Transitioning to Adult Cleft Care

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      Authors: Kenny Ardouin
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Kenny Ardouin grew up in East Sussex in the United Kingdom (UK), before moving to New Zealand, which he now considers home. Having been born with a cleft lip and palate, Kenny has a personal and professional dedication to the field. He served as CEO of the charitable organization Cleft New Zealand and recently completed 3.5 years as the Adult Services Manager for the Cleft Lip and Palate Association in the UK. Kenny works as a speech and language therapist, and is studying a Master’s degree part-time, focused on the psychological impact of speech differences in adolescents and adults born with a cleft. Kenny is also a radio broadcaster and editor and a freelance public speaker. In this heartfelt and informative invited editorial, Kenny reflects on how his treatment journey unfolded as a young adult and offers key learning points for all health professionals wanting to become more person-focused in their clinical practice.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-13T12:20:58Z
      DOI: 10.1177/10556656211043417
       
  • Does Early Secondary Alveolar Bone Grafting Influence Need for Additional
           Maxillary Advancement Procedures in Cleft Lip and Palate'

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      Authors: Kathlyn K. Powell, Paul Lewis, Rae Sesanto, Peter D. Waite
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo determine if secondary alveolar bone grafting (SABG) timing in patients with cleft lip and palate (CLP) influences the future need for additional maxillary advancement procedures, particularly Le Fort I osteotomy with rigid external distraction (RED).DesignRetrospective cohort study. Groups were separated by SABG timing: early mixed dentition (ages 68 years) or late mixed dentition (ages 9-11 years). The criterion for RED was negative overjet ≥8 mm, and sufficient dental development for RED.SettingSingle tertiary care institution.PatientsPatients with CLP that underwent SABG from 2010 to 2015. Exclusion criteria included syndromic conditions, SABG surgery at age >12 years, current age
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-13T12:19:58Z
      DOI: 10.1177/10556656211042789
       
  • Tessier 3 and 4 Clefts and Choanal Atresia: An Unusual Association'

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      Authors: Cristiano Tonello, Danilo Augusto Nery dos Passos Martins, Marco Antônio Ferraz de Barros Baptista, Felipe Mondelli, Nancy Mizue Kokitsu Nakata, Leonardo Bezerra Feitosa, Nivaldo Alonso
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionCraniofacial clefts are rare congenital anomalies that might involve both soft tissue and skeletal components. The association of Tessier cleft number 3 and 4 with choanal atresia appears to be unusual and only few clinical cases have been reported in published literature.ObjectivesReport a series of 13 cases of choanal atresia in patients with Tessier numbers 3 or 4 clefts and the literature review on this topic.MethodsA literature review was undertaken via PUBMED database before April 2020 addressing the association between Tessier numbers 3 or 4 clefts and choanal atresia. Retrospective chart review of patients diagnosed with both comorbidities at a tertiary hospital expertised in craniofacial anomalies.ResultsLiterature review yielded 10 studies describing the relationship between choanal atresia and Tessier 3 and 4 facial clefts. We identified 98 patients diagnosed with medial oro-ocular facial clefts (Tessier 3 and 4) and 119 with choanal atresia at our institution over a 20 years time period. Altogether, 13 individuals were diagnosed with both malformations, 3 patients with number 3 cleft, and 10 patients with number 4 cleft. It represents 13.26% of the cases.ConclusionThis study highlights the features of Tessier 3 and 4 facial clefts associated with choanal atresia. Although the publications regarding this association are very scarce, the authors present the largest series of cases of Tessier number 3 and 4 clefts with choanal atresia showing that association between these conditions could be not so unusual.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-13T12:18:57Z
      DOI: 10.1177/10556656211042172
       
  • Postoperative Use of Ketorolac Improves Pain Management and Decreases
           Narcotic Use Following Primary Cleft Palate Surgery

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      Authors: Samuel Dudley, Matthew Spence, Robert Frederick, Rosemary Stocks
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo study the efficacy and safety profile of ketorolac in cleft palate surgery.DesignRetrospective analysis of patients who underwent primary cleft palate surgery and received either postoperative ketorolac or opioids.SettingTertiary care children's hospital.Patients, ParticipantsEighty-nine patients enrolled who were all younger than 36 months of age, not dependent on a gastrostomy tube, with no history of bleeding disorders, and had undergone their primary cleft palate procedure by one specific surgeon between January 2010 and June 2019.Interventionsn/a.Main Outcome MeasureMorphine equivalent dose (MED), Face, Legs, Activity, Cry, Consolability (FLACC) score, length of stay (LOS), total oral intake (mL), total oral intake/LOS, and postoperative adverse events between ketorolac and no ketorolac groups.ResultsMED, FLACC score, and LOS were significantly lower in the ketorolac group compared to the no ketorolac group. One patient in the ketorolac group had a bleeding event.ConclusionsUse of ketorolac significantly decreased narcotic usage and pain scores as reported by the FLACC score. Moreover, postoperative bleeding was rare in both ketorolac and no ketorolac groups.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-13T12:17:46Z
      DOI: 10.1177/10556656211042168
       
  • Infant with Clefts Observation Outcomes Instrument (iCOO): A New Outcome
           for Infants and Young Children with Orofacial Clefts

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      Authors: Todd C. Edwards, Carrie L. Heike, Kathleen A. Kapp-Simon, Salene M. Jones, Brian G. Leroux, Laura P. Stueckle, Claudia Crilly Bellucci, Janine M. Rosenberg, Meredith Albert, Cassandra L. Aspinall, Donald L. Patrick
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveWe evaluated the measurement properties for item and domain scores of the Infant with Clefts Observation Outcomes Instrument (iCOO).DesignCross-sectional (before lip surgery) and longitudinal study (preoperative baseline and 2 days and 2 months after lip surgery).SettingThree academic craniofacial centers and national online advertisements.ParticipantsPrimary caregivers with an infant with cleft lip with or without cleft palate (CL  ±  P) scheduled to undergo primary lip repair. There were 133 primary caregivers at baseline, 115 at 2 days postsurgery, and 112 at 2 months postsurgery.Main Outcome Measure(s)Caregiver observation items (n = 61) and global impression of health and function items (n = 8) across eight health domains.ResultsMean age at surgery was 6.0 months (range 2.7-11.8 months). Five of eight iCOO domains have scale scores, with Cronbach’s alphas ranging from 0.67 to 0.87. Except for the Facial Skin and Mouth domain, iCOO scales had acceptable intraclass correlation coefficients (ICCs) ranging from 0.76 to 0.84. The internal consistency of the Global Impression items across all domains was 0.90 and had acceptable ICCs (range 0.76-0.91). Sixteen out of 20 (nonscale) items had acceptable ICCs (range 0.66-0.96). As anticipated, iCOO scores 2 days postoperatively were generally lower than baseline and scores 2 months postsurgery were consistent with baseline or higher. The iCOO took approximately 10 min to complete.ConclusionsThe iCOO meets measurement standards and may be used for assessing the impact of cleft-related treatments in clinical research and care. More research is needed on its use in various treatment contexts.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-13T12:16:56Z
      DOI: 10.1177/10556656211040307
       
  • The Impact of Social Determinants of Health in Facial and
           Craniomaxillofacial Reconstruction: Can We Do Better'

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      Authors: Sumun Khetpal, Daniel C. Sasson, Joseph Lopez, Derek M. Steinbacher, Arun K. Gosain
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Social determinants of health (SDOH) are integral to consider when delivering craniomaxillofacial and facial reconstructive care for patients. The American Cleft Palate-Craniofacial Association (ACPA) has instituted a formalized multidisciplinary care team model that recognizes such determinants and has aggregated patient-led organizations to strengthen patients’ education and support system. This review discusses the need for all surgeons engaged in facial and craniomaxillofacial reconstruction to consider SDOH in their practice. Additionally, we explore how factors such as race, insurance status, education level, cost, and access to follow-up care, impact surgical care for craniosynostosis, facial trauma, orthognathic surgery, head and neck cancer, and facial paralysis. We propose that the ACPA team model be applied to other societies that care for the broader scope of patients in need of facial and craniomaxillofacial reconstruction to strengthen the communication, collaboration, and standardization of care delivery that is personalized to the needs of each patient.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-13T12:11:28Z
      DOI: 10.1177/10556656211037510
       
  • Reading Development in Children With Nonsyndromic Cleft Palate With or
           Without Cleft Lip: Meta-analysis and Systematic Review

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      Authors: Hope Sparks Lancaster, Kari M. Lien, Jordan Haas, Paige Ellis, Nancy J. Scherer
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveWe conducted a meta-analysis and systematic review of literature comparing pre-reading and general reading in school-age children with nonsyndromic cleft palate with or without cleft lip (NSCP/L) to their peers without NSCP/L.MethodsOur literature search identified 1238 possible records. After screening we identified 11 samples for inclusion for systematic review and eight for meta-analysis. We compared 292 children with NSCP/L to 311 peers for 23 pre-reading effect sizes and 17 general reading effect sizes (EFg). We conducted a random-effects metaregression using robust variance estimation.ResultsOn average school-age children with NSCP/L scored lower on pre-reading (EFg = −0.36) and general reading measures (EFg = −0.38) compared to their peers. We conducted post-hoc analyses on phonological awareness and word decoding effect sizes; children with NSCP/L performed lower on phonological awareness (EFg = −0.22) and word decoding (EFg = −0.39) compared to their peers. There was weak evidence that hearing status and/or speech-language functioning might moderate reading development. There was limited evidence that age or socioeconomic status moderated reading development. However, samples did not consistently report several characteristics that were coded for this project.ConclusionsOur findings suggest that school-age children with NSCP/L have persistent reading problems. Further research is needed to explore reading development in children with NSCP/L, as well as the relationships among hearing, speech, language, and reading development.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-09-13T05:03:10Z
      DOI: 10.1177/10556656211039871
       
  • Quality of Life in Patients With Velopharyngeal Insufficiency in West
           China

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      Authors: Lidan Lu, Aipiziguli Yakupu, Yanhui Wu, Xiangnan Li, Pengxin Zhang, Guliziba Aihaiti, Hui Xu
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aimed to investigate the quality of life (QOL) of patients with cleft lip and palate and velopharyngeal insufficiency (VPI) in relation to sex, age, age at initial cleft lip surgery, and age at initial cleft palate surgery.DesignThis is a cross-sectional study.SettingThe study was conducted in a tertiary medical center.ParticipantsThe participants were caregivers of 72 patients with cleft lip and palate and VPI aged 4 to 20 years.Main Outcome Measure(s)Participants completed the Chinese version of the caregiver report of the VPI Effects on Life Outcomes (VELO) questionnaire. The Mann–Whitney U test was used to evaluate the patients’ sex, age, age at initial cleft lip repair, and age at initial cleft palate repair in relation to VELO total score and domains. Spearman correlation analysis was completed including all study variables. Associations between the study variables and the VELO total score were tested using a generalized linear mixed model.ResultsIn the univariate analysis, patients’ age and age at initial cleft palate surgery influenced the QOL of patients with VPI. There were no differences in the VELO total score or domains based on sex or age at first cleft lip surgery. In the generalized linear mixed model, patients older than 8 years had higher VELO total scores.ConclusionsBy caregiver report, the QOL of patients under age 8 years with VPI was lower than older patients. In addition, the caregiver impact domain was higher for parents of children who had their initial cleft palate surgery at age 2 years or younger.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-30T04:39:01Z
      DOI: 10.1177/10556656211034107
       
  • Novel GRHL3 Variants in a South African Cohort With Cleft Lip and Palate

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      Authors: Thirona Naicker, Chinyere C. Adeleke, Azeez Alade, Peter A. Mossey, Waheed A. Awotoye, Tamara D. Busch, Mary Li, Joy Olotu, Lord J. J. Gowans, Colleen Aldous, Azeez Butali
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe etiology of cleft palate (CP) is poorly understood compared with that of cleft lip with or without palate (CL ± P). Recently, variants in Grainyhead like transcription factor 3 (GRHL3) were reported to be associated with a risk for CP in European and some African populations including Nigeria, Ghana, and Ethiopia. In order to identify genetic variants that may further explain the etiology of CP, we sequenced GRHL3 in a South African population to determine if rare variants in GRHL3 are associated with the presence of syndromic or nonsyndromic CP.DesignWe sequenced the exons of GRHL3 in 100 cases and where possible, we sequenced the parents of the individuals to determine the segregation pattern and presence of de novo variants.SettingThe cleft clinics from 2 public, tertiary hospitals in Durban, South Africa (SA), namely Inkosi Albert Luthuli Central Hospital and KwaZulu-Natal Children's Hospital.Patients, participantsOne hundred patients with CL ± P and their parents.InterventionsSaliva samples were collected.Main outcome measuresTo ascertain the genetic variants in the GRHL3 gene in patients with CL ± P in SA.ResultsFive variants in GRHL3 were observed; 3 were novel and 2 were known variants. The novel variants were intronic variants (c.1062 + 77A>G and c.627 + 1G>A) and missense variant (p.Asp169Gly).ConclusionsThis study provides further evidence that variants in GRHL3 contribute to the risk of nonsyndromic CP in African populations, specifically, in the South African population.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-30T01:03:57Z
      DOI: 10.1177/10556656211038453
       
  • Ectomesenchymal Chondromyxoid Tumor: A Rare Association With an
           Asymmetrical Soft Palate Cleft

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      Authors: Sharan Naidoo, Gieljam J. Roode, Kurt W. Bütow, Shabnum Meer
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Ectomesenchymal chondromyxoid tumor (ECT) is a rare oral lesion first described by Smith et al. in 1995. These tumors are typically painless, slow growing and benign masses occurring predominantly on the anterior tongue dorsum. Prior to this seminal report, many ECTs may have been misdiagnosed due to the histological similarities with other lesions. Immunohistochemical stains aid in definitive diagnosis of an ECT. A total of 39 papers since published have reported 96 patients with ECT. Most lesions involve the anterior aspect of the tongue, with only 6 occurring in the posterior tongue and 2 involving the hard palate. ECTs are considered to develop from ectomesenchymal cells of neural crest cells that have migrated to the tongue during embryological development. This paper is of a rare case of ECT of the posterolateral tongue occurring in association with an unusual asymmetrical soft palate cleft. It is postulated that since the tongue develops before the formation of the soft palate, an ECT lesion occurring on the posterior aspect may have a causal contribution to the development of the soft palate cleft.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-30T01:01:46Z
      DOI: 10.1177/10556656211035029
       
  • Tessier Number 3 and 4 Clefts: Clinical Presentation and Associated Clefts
           in a South African Population

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      Authors: Abiola Omodan, Pamela Pillay, Lelika Lazarus, Kapil Satyapal, Anil Madaree
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionThe defects found in Tessier clefts number 3 and number 4 come in various forms in different patients. These variations have to a great extent affected not only documentation of these craniofacial defects but invariably their treatment and communication amongst craniofacial researchers. This study has not only documented the clinical presentation of these clefts in a South African population but has also incorporated the clinical presentation of Tessier clefts number 3 and 4 from different regions of the world.MethodsThe records of 8 patients, who had been treated for either Tessier clefts number 3 or 4, were reviewed and compared with 16 studies pulled from the literature systematically. The defects recorded as well as associated clefts and other congenital malformations were documented, and findings were compared.ResultsThe anatomical and clinical presentation of the patients was compared to the reviewed literature and the different parameters were documented. In addition, associated clefts were also recorded in the study—it was noted that the association pattern recorded in Tessier cleft number 4 in this study did not conform to its traditional counterpart.ConclusionThis study concluded that the clinical presentations of these clefts, however variable, seem to have a similar presentation worldwide. Additionally, associated clefts do not conform to the original Tessier classification system and therefore it is imperative for these patterns to be clearly mapped out.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-20T11:24:17Z
      DOI: 10.1177/10556656211036306
       
  • Simultaneous Fat Injection for Nasal Contouring in Orthognathic Patients

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      Authors: Daniel Lonic, Yen-Chang Hsiao, Jung-Ju Huang, Chun-Shin Chang, Jyh-Ping Chen, Paul I. Heidekrueger, Andreas Kehrer, Lukas Prantl, Lun-Jou Lo, Yu-Ray Chen
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:Augmentation rhinoplasty with autologous fat grafting is a useful procedure to meet the demand for facial harmonization in the Asian population. We used this procedure during orthognathic surgery to address inadequate dorsum projection. This prospective study was conducted to determine the fat retention rate in patients undergoing simultaneous autologous fat injection augmentation rhinoplasty and orthognathic surgery.Methods:Nineteen patients were treated with simultaneous bimaxillary orthognathic surgery and autologous fat grafting of the nasal dorsum and tip. The paired t test was used to compare the nasal volumes before and at least 6 months after surgery measured by 3-dimensional computer tomography scans. All measurements were performed twice by the same evaluator at least 2 weeks apart for intrarater consistency.Results:Seventeen patients completed the study. The volume means before and after surgery were 22.3 ± 4.6 cm3 and 23.3 ± 4.7 cm3, respectively, with a mean difference of 1.0 ± 0.3 cm3 (P < .001). The mean retention rate was calculated to be 50.5% ± 7.0% (range: 40.5%-64.7%). Intrarater consistency was high with a Cronbach α of .97 (P < .001) and .98 (P < .001), respectively.Conclusion:This prospective study provides objective graft retention measurements for fat injection augmentation rhinoplasty combined with orthognathic surgery. All patients were satisfied with the results and no complications or additional morbidity was noted in the postoperative course. We consider this procedure to be a safe, reliable, and powerful adjunct to improve the aesthetic results of orthognathic surgery.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-20T09:07:22Z
      DOI: 10.1177/10556656211026476
       
  • Investigation of Genetic Causes in a Developmental Disorder:
           Oculoauriculovertebral Spectrum

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      Authors: Naz Güleray, Can Koşukcu, Sümeyra Oğuz, Gizem Ürel Demir, Ekim Z. Taşkıran, Pelin Özlem Şimşek Kiper, Gülen Eda Utine, Yasemin Alanay, Koray Boduroğlu, Mehmet Alikaşifoğlu
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveOculoauriculovertebral spectrum (OAVS) is a genetically and clinically heterogeneous disorder that occurs due to a developmental field defect of the first and second pharyngeal arches. Even though recent whole exome sequencing studies (WES) have led to identification of several genes associated with this spectrum in a subset of individuals, complete pathogenesis of OAVS remains unsolved. In this study, molecular genetic etiology of OAVS was systematically investigated.Design/Setting/PatientsA cohort of 23 Turkish patients with OAVS, referred to Hacettepe University Hospital, Department of Pediatric Genetics from 2008 to 2018, was included in this study. Minimal diagnostic criteria for OAVS were considered as unilateral microtia or hemifacial microsomia with preauricular skin tag. The cohort was clinically reevaluated for craniofacial and extracranial findings. Molecular etiology was investigated using candidate gene sequencing following copy number variant (CNV) analysis. WES was also performed for 2 of the selected patients.ResultsPatients in the study cohort presented similar demographic and phenotypic characteristics to previously described patients in the literature except for a higher frequency of bilaterality, cardiac findings, and intellectual disability/developmental delay. CNV analysis revealed a possible genetic etiology for 3 patients (13%). Additional WES in 1 of the 2 patients uncovered a novel heterozygous nonsense variant in Elongation factor Tu GTP-binding domain-containing 2 (EFTUD2) causing mandibulofacial dysostosis with microcephaly (MFDM), which clinically overlaps with OAVS.ConclusionDetailed clinical evaluation for any patient with OAVS is recommended due to a high rate of accompanying systemic findings. We further expand the existing genetic heterogeneity of OAVS by identifying several CNVs and a phenotypically overlapping disorder, MFDM.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-19T12:34:53Z
      DOI: 10.1177/10556656211038115
       
  • Tarsoconjunctival-Skin Flap as Another Option in Correcting Ectropion for
           Oro-Ocular Cleft Reconstruction: Report of 2 Cases

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      Authors: Magda R. Hutagalung, Indri L. Putri, Siti I. Wahdini, Ishandono Dachlan, Lobredia Zarasade
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Facial clefts are rare facial anomalies. Among them, oro-ocular cleft remains a challenging malformation due to ectropion of the inferior palpebra which can constitute an emergency when the patient's globe is exposed leading to exposure keratitis and blindness. Here we report surgical procedures to correct lower eyelid ectropion using tarsoconjunctival-skin flap performed on 2 cases. In conclusion, tarsoconjunctival-skin flap can be provided to effectively correct lower eyelid ectropion and is a better option when nose correction is not involved simultaneously.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-19T12:33:30Z
      DOI: 10.1177/10556656211036620
       
  • Continuous Multidisciplinary Care for Patients With Orofacial
           Clefts—Should the Follow-up Interval Depend on the Cleft Entity'

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      Authors: Anna K. Sander, Elisabeth Grau, Anita Kloss-Brandstätter, Rüdiger Zimmerer, Michael Neuhaus, Alexander K. Bartella, Bernd Lethaus
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe multidisciplinary follow-up of patients with cleft lip with or without palate (CL/P) is organized differently in specialized centers worldwide. The aim of this study was to evaluate the different treatment needs of patients with different manifestations of CL/P and to potentially adapt the frequency and timing of checkup examinations accordingly.DesignWe retrospectively analyzed the data of all patients attending the CL/P consultation hour at a tertiary care center between June 2005 and August 2020 (n = 1126). We defined 3 groups of cleft entities: (1) isolated clefts of lip or lip and alveolus (CL/A), (2) isolated clefts of the hard and/or soft palate, and (3) complete clefts of lip, alveolus and palate (CLP). Timing and type of therapy recommendations given by the specialists of different disciplines were analyzed for statistical differences.ResultsPatients with CLP made up the largest group (n = 537), followed by patients with cleft of the soft palate (n = 371) and CL ± A (n = 218). There were significant differences between the groups with regard to type and frequency of treatment recommendations. A therapy was recommended in a high proportion of examinations in all groups at all ages.ConclusionAlthough there are differences between cleft entities, the treatment need of patients with orofacial clefts is generally high during the growth period. Patients with CL/A showed a similarly high treatment demand and should be monitored closely. A close follow-up for patients with diagnosis of CL/P is crucial and measures should be taken to increase participation in follow-up appointments.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-19T12:32:37Z
      DOI: 10.1177/10556656211035253
       
  • Description of Total Population Hospital Admissions for Treacher Collins
           Syndrome in Australia

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      Authors: Mohammed Junaid, Linda Slack-Smith, Kingsley Wong, Gareth Baynam, Hanny Calache, Timothy Hewitt, Helen Leonard
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo describe patterns and demographic characteristics of total-population hospital admissions with a diagnosis of Treacher Collins syndrome (TCS) in Australia.Data SourcePopulation summary data for inpatient hospitals admissions (public and private) with a principal diagnosis of TCS (ICD10-AM-Q87.04) were obtained from the Australian Institute of Health and Welfare National Hospital Morbidity Database for a 11-year period (2002-2013).Main Outcome MeasuresThe primary outcome was hospital separation rate (HSR), calculated by dividing the number of hospital separations by estimated resident population per year. Trends in HSR s adjusted for age and sex were investigated by negative binomial regression presented as annual percent change and the association of rates with age and sex was expressed as incidence rate ratio.ResultsIn 244 admissions identified, we observed an increase of 4.55% (95% confidence interval [CI] −1.78, 11.29) in HSR's over the 11-year period. Rates were higher during infancy (1.87 [95% CI 1.42, 2.42]), declining markedly with increasing age. The average length of hospital stay was 6.09 days (95% CI 5.78, 6.40) per episode, but longer for females and infants.ConclusionsFindings indicate an increase in hospitalization rates, especially among infants and females which potentially relates to early airway intervention procedures possibly influenced by sex specific-disease severity and phenotypic variability of TCS. Awareness of the TCS phenotype and improved access to genetic testing may support more personalized and efficient care. Total-population administrative data offers a potential to better understand the health burden of rare craniofacial diseases.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-19T03:19:18Z
      DOI: 10.1177/10556656211036618
       
  • Early Adverse Events Following Pediatric Mandibular Advancement: Analysis
           of the ACS NSQIP-Pediatric Database

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      Authors: Kevin J. Carlson, Suhas R. Bharadwaj, William M. Dougherty, Eric J. Dobratz
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aims to assess early adverse events and patient factors associated with complications following mandible distraction osteogenesis (MDO).Materials and MethodsThe American College of Surgeons National Surgical Quality Improvement Program-Pediatric (NSQIP-Pediatric) database, years 2012 to 2019, was queried for patients undergoing mandible advancement via relevant Current Procedural Terminology and postoperative diagnosis codes. Thirty-day adverse events and co-morbidities are assessed.ResultsA total of 208 patients were identified with 17.3% (n = 36) experiencing an adverse event, reoperation (n = 14), and readmission (n = 11) being most common. Patients 28 days (30.2% vs 22.2%; P = .47) and those weighing  ≤ 4 kg and >4 kg (31.7% vs 11.5%; P = .063) did not reach statistical significance.ConclusionsAdverse events following mandible advancement are relatively common, though often minor. In our analysis of the NSQIP-Pediatric database, neonatal age ( ≤ 28 days) or weight  ≤ 4 kg did not result in a statistically significant increase in complications among patients less than 1 year of age. Providers should consider early intervention in patients who may benefit from MDO.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-18T11:38:37Z
      DOI: 10.1177/10556656211037852
       
  • Three-Dimensional Analysis of Definitive Secondary Unilateral Cleft
           Rhinoplasty Using Cartilage Graft

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      Authors: Thinnapat Hantawornchaikit, Raweewan Arayasantiparb, Kumar KC, Kiatanant Boonsiriseth
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThree-dimensional assessment of nasal morphology in patients with unilateral cleft lip nose treated by cartilage graft augmentation.DesignRetrospective study.Patients and InterventionThirteen patients with unilateral cleft lip nose underwent definitive secondary rhinoplasty and postsurgical changes were examined using a three-dimensional (3D) laser scan.Main Outcome MeasureNasal dorsum length, nasal tip, alar width, and alar base width in frontal view; nasion depth, nasal tip projection, nasal dorsal angle, and nasal tip angle in lateral view; nostril width, nostril height, and nasal tip height in basal view were measured at preoperative (T1: within 6 months), short follow-up (T2: 2-10 weeks), and long follow-up (T3: 9-14 months).ResultsA significant change in alar width, alar base width, nostril width, and nostril height at the cleft side, nasal dorsum length, nasion depth, nasal tip projection, and nasal tip height was observed from T1 to T3 follow-up after rhinoplasty (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-17T11:53:20Z
      DOI: 10.1177/10556656211034099
       
  • Nasal Reconstruction of a Frontonasal Dysplasia via Septal L-Strut
           Reconstruction Using Costal Cartilage

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      Authors: Yong Bae Kim, Seung Min Nam, Eun Soo Park, Chang Yong Choi, Han Gyu Cha, Jun Hyun Kim
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveFrontonasal dysplasia (FND) is a rare congenital condition. Its major features include hypertelorism, a large and bifid nasal tip, and a broad nasal root. We present our technique of septal L-strut reconstruction using costal cartilage.DesignRetrospective review from June 2008 and August 2017.MethodsUnder general anesthesia, 6 patients with FND underwent septal reconstruction using costal cartilage via open rhinoplasty. We reconstructed the nasal and septal cartilaginous framework by placing columellar struts and cantilever-type grafts.ResultsThe patients ranged in age from 6 to 13 years old. All were female. The follow-up period ranged from 8 months to 2 years; we encountered no postoperative complications (infection, nasal obstruction, or recurrence). All patients were satisfied with their nasal appearance.ConclusionsAlthough the results were not entirely satisfactory from an esthetic point of view, we found that FND can be treated via septal reconstruction with costal cartilage and that the clinical outcomes are reliable and satisfactory. Our approach is a useful option for FND patients.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-17T10:41:38Z
      DOI: 10.1177/10556656211036614
       
  • Anterior “W” Tongue Reduction for Macroglossia in
           Beckwith-Wiedemann Syndrome

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      Authors: Bar Y. Ainuz, Emily L. Geisler, Rami R. Hallac, Jeyna K. Perez, James R. Seaward, Alex A. Kane
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionMacroglossia occurs in 80% to 99% of patients with Beckwith-Wiedemann syndrome (BWS) and a variety of surgical techniques for tongue reduction are offered by surgeons. The purpose of this study is to evaluate the postoperative outcomes of the anterior “W” tongue reduction technique in patients with BWS.MethodsA retrospective review was conducted of all patients diagnosed with BWS that underwent an anterior “W” tongue reduction for macroglossia in the past 7 years, performed by 2 surgeons. Demographics, procedural characteristics, perioperative outcomes, and complications were assessed.ResultsA total of 19 patients met inclusion criteria consisting of 8 male and 11 female patients. The mean age at the time of surgery was 405 days, mean surgeon operating time was 1.06 h, and mean length of follow-up was 467 days. Postoperative oral competence was observed in 100% of patients. There was no reported history of sleep apnea or airway compromise. Speech delay was seen in 4 patients pre- and postoperatively. Feeding issues decreased from 7 patients preoperatively to 1 patient postoperatively. Preoperative prevalence of class III malocclusion (53%) and isolated anterior open bite (26%) decreased postoperatively to 37% and 16%, respectively. The only reported complications were superficial tip wound dehiscence in 3 patients treated with nystatin antifungal therapy. None of the patients required revisional surgery.ConclusionPatients treated with the anterior “W” tongue reduction technique had low rates of perioperative complications and significant improvements in oral competence. Anterior “W” tongue reduction is safe and effective for the correction of macroglossia in patients with BWS.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-17T10:40:39Z
      DOI: 10.1177/10556656211036607
       
  • A Guide to Bilateral Cleft Lip Markings: An Anthropometric Study of the
           Normal Cupid's Bow

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      Authors: Yew L. Loo, Aaron C. Van Slyke, Pragaash Shanmuganathan, Robert Reitmaier, David K. Chong
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe aim of this study was to define the ratio of the heights of the vermilion between the peaks and trough of the Cupid’s bow and hence assist in defining the point of closure on the lateral lip element to achieve a balanced Cupid's bow in bilateral lip repair based on our findings.DesignThis study is a retrospective observational study of the anthropometrics of the upper lip's Cupid’s bow. Three-dimensional (3D) images of toddlers between 2009 and 2016 were extracted from a normative 3D image database of toddlers at the Royal Children's Hospital, Melbourne.PatientsParticipants from the normative 3D image database at the age of 1 year were studied. This excluded any patients with prior trauma or surgical intervention of the nasolabial region.InterventionLandmarks measured were right and left crista philtri (cphR and cphL), labial superius (ls), stomion (sto), right and left chelion, and labial fissure (lf).OutcomeVermilion height of the right peak, trough, and left peak of the Cupid’s bow was calculated by analyzing the vertical linear distance between cphR–lf, ls–sto, and cphL–lf. The ratio between the median and paramedian heights were recorded.ResultsThe paramedian height of the upper lip vermilion is consistently greater than the midline height. There was no significant sexual dysmorphism between ratio of paramedian to midline height on the right (P = .538) and left (P = .410).ConclusionWe describe an anthropometric observation of the vermilion relationship at the Cupid’s bow and define a specific lateral lip marking for bilateral cleft lip repair based on our anthropometric findings.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-17T10:39:18Z
      DOI: 10.1177/10556656211036329
       
  • Micro-Computed Tomography-Based Three-Dimensional Anatomical Structure of
           the Region Around the Pterygoid Hamulus

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      Authors: Jiuli Zhao, Hengyuan Ma, Yongqian Wang, Tao Song, Chanyuan Jiang, Di Wu, Ningbei Yin
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivePalatoplasty would involve the structures around the pterygoid hamulus. However, clinicians hold different opinions on the optimal approach for the muscles and palatine aponeurosis around the pterygoid hamulus. The absence of a consensus regarding this point can be attributed to the lack of investigations on the exact anatomy of this region. Therefore, we used micro-computed tomography to examine the anatomical structure of the region surrounding the pterygoid hamulus.DesignCadaveric specimens were stained with iodine–potassium iodide and scanned by micro-computed tomography to study the structures of the tissues, particularly the muscle fibers. We imported Digital Imaging and Communications in Medicine images to Mimics to reconstruct a 3-dimensional model and simplified the model.ResultsThree muscles were present around the pterygoid hamulus, namely the palatopharyngeus (PP), superior constrictor (SC), and tensor veli palatini (TVP). The hamulus connects these muscles as a key pivot. The TVP extended to the palatine aponeurosis, which bypassed the pterygoid hamulus, and linked the PP and SC. Some muscle fibers of the SC originated from the hamulus, the aponeurosis of which was wrapped around the hamulus. There was a distinct gap between the pterygoid hamulus and the palatine aponeurosis. This formed a pulley-like structure around the pterygoid hamulus.ConclusionsTransection or fracture of the palatine aponeurosis or pterygoid hamulus, respectively, may have detrimental effects on the muscles around the pterygoid hamulus, which play essential roles in the velopharyngeal function and middle ear ventilation. Currently, cleft palate repair has limited treatment options with proven successful outcomes.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-17T10:38:19Z
      DOI: 10.1177/10556656211036302
       
  • Furlow Palatoplasty for Velopharyngeal Dysfunction Management: Auditing
           and Predicting Outcomes

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      Authors: Krishnamurthy Bonanthaya, Jazna Jalil, Aparna V. Sasikumar, Pritham N. Shetty
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe Furlow palatoplasty is a proven procedure in the management of secondary velopharyngeal dysfunction in patients with cleft palate. But the selection of cases, the degree of clinical success, and the preoperative predictors of the same are less established. This study is an effort to retrospectively look at outcomes, in a large series of velopharyngeal dysfunction cases treated with the Furlow palatoplasty alone.DesignRetrospective analysis of preoperative and postoperative speech and videofluoroscopic data.SettingTertiary care center.Patients/ParticipantsNinety-two patients who were diagnosed with velopharyngeal dysfunction post primary cleft palate repair.InterventionsFurlow palatoplasty for velopharyngeal dysfunction post primary cleft palate repair.Main outcome measuresVariables analyzed were perceptual speech parameters and, closure ratios obtained from lateral video-fluoroscopic images.ResultsOverall, 81.5% had postoperative improvements in their lateral video-fluoroscopic parameters, 63% improved their nasality scores, and 65.2% had improved speech intelligibility. A simple linear regression was done to predict the postoperative closure ratio. Preoperative closure ratio, hypernasality (moderate and severe), and audible nasal air emission are predictors for postoperative closure ratio.ConclusionsThe Furlow palatoplasty alone led to complete resolution, or significant improvement of velopharyngeal dysfunction in a majority of patients, despite the cohort having a wide range of severity in terms of degree of dysfunction. The predictive formula will be validated in a further study.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-17T10:37:06Z
      DOI: 10.1177/10556656211035914
       
  • The Current State of Cleft Care in Sub-Saharan Africa: A Narrative Review

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      Authors: James R. Wester, Joshua P. Weissman, Narainsai K. Reddy, Emily S. Chwa, Arun K. Gosain
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo identify practices and limitations of cleft care in sub-Saharan Africa (SSA).DesignA retrospective narrative nonsystematic literature review was performed.SettingLiterature exploring the management practices of cleft lip and/or palate across regions in SSA was included.ParticipantsFull text case reports, retrospective studies, prospective studies, clinical trials, and review articles written and published in English between 1966 and February 1, 2021, were included in this analysis utilizing PubMed, MEDLINE, EMBASE, and Google scholar databases.Main Outcome MeasuresQualitative themes identified in analysis were clinical practice patterns, current infrastructure and limitations of cleft repair, training and interdisciplinary teams, economic analyses, and international partnerships.ResultsSignificant barriers to care identified in SSA include lack of hospital resources, craniofacial training, access to multidisciplinary specialists, and public awareness. These problems make the entire care journey difficult for patients. Increasing public education has the power to diminish late presentations to hospitals. Providing adequate hospital resources and craniofacial training through international and organizational partnerships can ensure that more patients will receive care. Increasing the availability and number of multidisciplinary specialists is crucial to follow up care which aims at improving functional outcomes.ConclusionThis narrative review highlights current practices and limitations in cleft care, emphasizing the importance of effective and timely repair of clefts in SSA. Targeted efforts aimed at establishing sustainable infrastructure for cleft care in SSA can have significant individual and community health and economic benefits.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-16T12:47:15Z
      DOI: 10.1177/10556656211038183
       
  • A Clinical Report Utilizing the VITOM 3D® Microvideoscope for Cleft
           Palate Repair

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      Authors: Peter J. Meier, Douglas G. Copson, David A. Gillett
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Cleft palate surgery has traditionally presented numerous problems for cleft surgeons including ergonomics, limited visual fields restricting the opportunity for demonstration and teaching. Additionally, the move toward online teaching means the ability to record or livestream video is paramount. The following report of eight cleft palate repairs highlights the novel use of the Vitom 3D® microvideoscope as an innovative technique for cleft palate repair with our early experience demonstrating significant ergonomic and teaching benefits.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-16T12:44:54Z
      DOI: 10.1177/10556656211036608
       
  • Comparative Assessment of Anxiety, Pain, and Discomfort During Rapid
           Maxillary Expansion Using Two Different Activation Protocols in Patients
           With Cleft Lip and Palate

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      Authors: Dipjyoti Baruah, Shweta Nagesh, Hiteswar Sarma, Navraj Mattu
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe aim of this study was to compare and assess the pain perception, anxiety, and discomfort between two different rapid maxillary expansion (RME) protocols in patients with unilateral cleft lip and palate (UCLP).DesignThis is a prospective study.SettingThe study was done in a comprehensive cleft care center.Patien and ParticipantsThe sample included 26 patients between ages 6 and 9 years with a history of repaired UCLP.InterventionsThe patients were randomly allotted into 2 groups—Group A and Group B. Group A received 1 RME activation per day and Group B received 2 activations per day. The participants after RME activation completed a self-report questionnaire and ranked the pain and anxiety levels using Faces pain scale and Hamilton anxiety scale, respectively. The evaluation was done every day for 1 week.Main Outcome MeasureThe Faces Pain Scale, Hamilton Anxiety Scale, and self-report questionnaires assessed the pain, anxiety, and discomfort experienced by the patients after 2 different types of RME activation protocols.ResultsGroup B experienced significantly higher pain in the molars than Group A (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-16T12:44:15Z
      DOI: 10.1177/10556656211033105
       
  • Predictive Factors for Velopharyngeal Insufficiency Following Primary
           Cleft Palate Repair

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      Authors: Brady J. Anderson, Kasra N. Fallah, Austin A. Lignieres, Joseph K. Moffitt, Kim-Loan Luu, Alfredo Cepeda, Irene L. Doringo, Phuong D. Nguyen, John F. Teichgraeber, Matthew R. Greives
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Velopharyngeal insufficiency (VPI) remains a known complication of primary palatoplasty. We sought to identify factors associated with the incidence of VPI and create a predictive model for VPI development in our population.Design:A single-institution, retrospective review.Setting:Multidisciplinary clinic in a tertiary academic institution.Patients:A total of 453 consecutive patients undergoing primary palatoplasty from 1999 to 2016 were reviewed. Inclusion required follow-up past age 5. Patients who were non-verbal, and thus unable to undergo speech evaluation, were excluded.Main Outcome Measures:Primary outcome was VPI, defined as revision palatoplasty or recommendation by speech-language pathology.Results:Of 318 patients included, 179 (56%) were male. Median age at primary repair was 1.0 years (0.9-1.1) with a median age of 8.8 years at last follow-up. One hundred nineteen (37%) patients developed VPI at a median age of 5.0 years (3.8-6.5). Higher rates were seen with posterior fistula (65% vs 14%, P
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-16T09:08:01Z
      DOI: 10.1177/10556656211026861
       
  • Core Outcome Set for Reporting Outcomes of Interventions for
           Velopharyngeal Dysfunction: Final Results of the COS-VPD Initiative

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      Authors: Catherine de Blacam, Adriane L. Baylis, Richard E. Kirschner, Susan Smith, Debbie Sell, Kathleen C.Y. Sie, Helen E. Harris, David J.A. Orr
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo date, the recording of outcomes of interventions for velopharyngeal dysfunction (VPD) has not been standardized. This makes a comparison of results between studies challenging. The aim of this study was to develop a core outcome set (COS) for reporting outcomes in studies examining the management of VPD.DesignA two-round Delphi consensus process was used to develop the COS.Patients, ParticipantsThe expert Delphi panel comprised patients and caregivers of patients with VPD, surgeons and speech and language therapists specializing in cleft palate, and researchers with expertise in VPD.InterventionsA long list of outcomes was derived from the published literature. In each round of a Delphi survey, participants were asked to score outcomes using the Grading of Recommendations, Assessment, Development, and Evaluations scale of 1 to 9, with 1 to 3 labeled “not important,” 4 to 6 labeled “important but not critical,” and 7 to 9 labeled “critical.”Main outcome measureConsensus criteria were specified a priori. Outcomes with a rating of 75% or more of the panel rating 7 to 9 and 25% or fewer rating 1 to 3 were included in the COS.ResultsA total of 31 core outcomes were identified from the Delphi process. This list was condensed to combine topic areas to produce a final COS of 10 outcomes, including both processes of care and patient-reported outcomes that should be considered for reporting in future studies of VPD.ConclusionsImplementation of the COS-VPD will facilitate consistency of outcomes data collection and comparison of results across studies.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-16T04:34:52Z
      DOI: 10.1177/10556656211035026
       
  • Phenotype Analysis and Genetic Study of Chinese Patients With Treacher
           Collins Syndrome

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      Authors: Meng Lu, Bin Yang, Zixiang Chen, Haiyue Jiang, Bo Pan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe aim of this study was to confirm the pathogenic variants, explore the genotype–phenotype correlation and characteristics of Chinese patients with Treacher Collins syndrome (TCS).DesignClinical details of 3 TCS family cases and 2 sporadic cases were collected and analyzed. Whole-exome sequencing and Sanger sequencing were conducted to detect causative variants.SettingTertiary clinical care.PatientsThis study included 8 patients clinically diagnosed with TCS who were from 3 familial cases and 2 sporadic cases.Main Outcome MeasuresWhen filtering the database, variants were saved as rare variants if their frequency were less than 0.005 in the 1000 Genomes Project Database, the Exome Aggregation Consortium (ExAC) browser, and the Novogene database, or they would be removed as common ones. The pathogenic variants identified were verified by polymerase chain reaction. The sequencing results were analyzed by Chromas 2.1 software.ResultsTwo novel pathogenic variants (NM_000356.3: c.537del and NM_000356.3: c.1965_1966dupGG) and 2 known pathogenic variants (NM_000356.3: c.1535del, NM_000356.3: c.4131_4135del) were identified within TCOF1 which are predicted to lead to premature termination codons resulting in a truncated protein. There was a known missense SNP (NM_015972.3: c.139G>A) within POLR1D. No phenotype–genotype correlation was observed. Instead, these 8 patients demonstrated the high genotypic and phenotypic heterogeneity of TCS.ConclusionsThis study expands on the pathogenic gene pool of Chinese patients with TCS. Besides the great variation among patients which is similar to international reports, Chinese patients have their own characteristics in clinical phenotype and pathogenesis mutations.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-16T03:22:43Z
      DOI: 10.1177/10556656211037509
       
  • Genome-Wide Scan for Parent-of-Origin Effects in a sub-Saharan African
           Cohort With Nonsyndromic Cleft Lip and/or Cleft Palate (CL/P)

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      Authors: Lord J. J. Gowans, Carissa L. Comnick, Peter A. Mossey, Mekonen A. Eshete, Wasiu L. Adeyemo, Thirona Naicker, Waheed A. Awotoye, Aline Petrin, Chinyere Adeleke, Peter Donkor, Tamara D. Busch, Olutayo James, Mobolanle O. Ogunlewe, Mary Li, Joy Olotu, Mohaned Hassan, Oluwole A. Adeniyan, Solomon Obiri-Yeboah, Fareed K. N. Arthur, Pius Agbenorku, Alexander A. Oti, Olubukola Olatosi, Olawale O. Adamson, Azeez A. Fashina, Erliang Zeng, Mary L. Marazita, Adebowale A. Adeyemo, Jeffrey C. Murray, Azeez Butali
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveNonsyndromic cleft lip and/or cleft palate (NSCL/P) have multifactorial etiology where genetic factors, gene–environment interactions, stochastic factors, gene–gene interactions, and parent-of-origin effects (POEs) play cardinal roles. POEs arise when the parental origin of alleles differentially impacts the phenotype of the offspring. The aim of this study was to identify POEs that can increase risk for NSCL/P in humans using a genome-wide dataset.MethodsThe samples (174 case-parent trios from Ghana, Ethiopia, and Nigeria) included in this study were from the African only genome wide association studies (GWAS) that was published in 2019. Genotyping of individual DNA using over 2 million multiethnic and African ancestry-specific single-nucleotide polymorphisms from the Illumina Multi-Ethnic Genotyping Array v2 15070954 A2 (genome build GRCh37/hg19) was done at the Center for Inherited Diseases Research. After quality control checks, PLINK was employed to carry out POE analysis employing the pooled subphenotypes of NSCL/P.ResultsWe observed possible hints of POEs at a cluster of genes at a 1 mega base pair window at the major histocompatibility complex class 1 locus on chromosome 6, as well as at other loci encompassing candidate genes such as ASB18, ANKEF1, AGAP1, GABRD, HHAT, CCT7, DNMT3A, EPHA7, FOXO3, lncRNAs, microRNA, antisense RNAs, ZNRD1, ZFAT, and ZBTB16.ConclusionFindings from our study suggest that some loci may increase the risk for NSCL/P through POEs. Additional studies are required to confirm these suggestive loci in NSCL/P etiology.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-08-12T02:43:38Z
      DOI: 10.1177/10556656211036316
       
  • Nasopharyngeal Tube and Functional Treatment in Pierre Robin Sequence: A
           Tertiary Clinical Experience From 150 Cases

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      Authors: Fabian Blanc, Inge Harrewijn, Claire Duflos, Frederica Maggiulli, Guillaume Captier
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:To describe the initial care practices for children with Pierre Robin sequence (PRS) and analyze the factors predicting the severity of the obstruction breathing disorders.Design:A retrospective single-center study of 150 children with PRS.Setting:Single tertiary care center, Regional Competence Center for the diagnosis and treatment of PRS.Patients:A total of 150 children with PRS consecutively followed between 1986 and 2017. Group 1 comprises children without specific respiratory management; group 2, children requiring prone positioning to alleviate their respiratory distress symptoms; and group 3, children requiring nasopharyngeal airway tube (NT) or nonconservative surgical treatment.Main outcome measures:Evolution and results of the initial treatment of PRS.Results:Forty-two percent (n = 63) were attributed to group 1, 39% (n = 50) to group 2, and 19% (n = 29) to group 3. Preterm birth, birth weight, or associated congenital malformations were not significantly different between the groups. However, the age of exclusive oral feeding was significantly different: 1 day (quartiles: 0-3) for group 1; 11 days (quartiles: 1-28) for group 2; 39 days (quartiles: 19-111) for group 3 (P < .0001). Considering the NT, its use relieves the upper airway obstruction, assessed by a respiratory polygraphy, in 14 children.Conclusions:Nasopharyngeal airway tube has become our major first-line treatment, avoiding more complex procedures in most of the cases. The achievement of exclusive oral feeding seems to be a good predictor of the severity of respiratory symptoms in PRS.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-27T09:58:16Z
      DOI: 10.1177/10556656211031105
       
  • Emotional and Behavioral Problems in Children With a Cleft Lip With or
           Without Palate or an Infantile Hemangioma

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      Authors: Marije van Dalen, Mireille M. Hermans, Willem H. Leemreis, Vivian Kraaij, Peter C. J. De Laat, Suzanne G. M. A. Pasmans, Sarah L. Versnel, Maarten J. Koudstaal, Manon H. J. Hillegers, Elisabeth M. W. J. Utens, Jolanda M. E. Okkerse
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Life can be challenging for children with a visible difference due to a medical condition, and they might be at risk for emotional and behavioral problems. This study examines emotional and behavioral problems in children with a cleft lip with or without palate (CL ± P) or an infantile hemangioma (IH) in relation to the visibility of the condition, the presence of additional condition-related problems, and parental affect.Setting:This cross-sectional study took place in an academic medical hospital in the Erasmus MC Sophia Children’s Hospital, the Netherlands.Participants:A total of 309 parents (mean age = 40.34, 44.00% male) of 182 children with CL ± P and 48 parents (mean age = 39.21, 37.50% male) of 33 children with an IH completed questionnaires. Children were 1.5 to 12 years old.Results:Parents reported fewer child emotional and behavioral problems compared to normative data. Problems reported were mainly related to learning difficulties and parent gender, while visibility of the condition had no significant influence. Parental negative affect was related to child internalizing problems. Parental positive affect was not related to any of the outcome measures.Conclusions:Parents reported fewer problems for their children compared to normative data. This is inconsistent with previous research, showing similar or worse scores for these children compared to peers. Our findings may be explained by a protective parenting style, a response shift in parents, or problems developing at a later point in life.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-23T08:59:40Z
      DOI: 10.1177/10556656211031411
       
  • Histologic Aspect of the Curved Vomerine Mucosa in Cleft Lip and Palate

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      Authors: Benito K. Benitez, Andrzej Brudnicki, Prasad Nalabothu, Jeannette A. von Jackowski, Elisabeth Bruder, Andreas Albert Mueller
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:Common surgical techniques aim to turn the entire vomerine mucosa with vomer flaps either to the oral side or to the nasal side. The latter approach is widely performed due to the similarity in color to the nasal mucosa. However, we lack a histologic description of the curved vomerine mucosa in cleft lip and palate malformations.Methods:We histologically examined an excess of curved vomerine mucosa in 8 patients using hematoxylin–eosin, periodic acid–Schiff, Elastin van Gieson, and Alcian blue stains. Tissue samples were obtained during surgery at 8 months of age.Results:Our histological analysis of the mucoperiosteum overlying the curved vomer revealed characteristics consistent with those of an oral mucosa or a squamous metaplasia of the nasal mucosa, as exhibited by a stratified squamous epithelium containing numerous seromucous glands. Some areas showed a palisaded arrangement of the basal cells compatible with metaplasia of respiratory epithelium, but no goblet cells or respiratory cilia were identified. Abundant fibrosis and rich vascularity were present.Conclusion:The vomer mucosa showed no specific signs of nasal mucosa. These findings should be considered in presurgical cleft orthopedics and palatal surgery for further refinement. Shifting the vomer mucosa according to a fixed physiologic belief should not overrule other important aspects of cleft repair such as primary healing and establishing optimal form and function of palatal roof and nasal floor.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-22T09:29:41Z
      DOI: 10.1177/10556656211031419
       
  • Reconstruction of Congenital Arhinia With Stereolithographic Modeling:
           Case Correlate and Literature Review

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      Authors: Lucas M. Harrison, Spencer R. Anderson, Kelly E. Spiller, Kaitlynne Y. Pak, Steven P. Schmidt, Salim N. Mancho
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Complete congenital arhinia is a rare defect of embryogenesis leading to the absence of the external nose and airway. We report our novel multistaged reconstructive approach and literature review. Nasal methyl methacrylate prosthesis was created from a stereolithographic model for use as a temporary prosthesis and tissue expander. Lefort 1 with cannulization was utilized for midface advancement and airway formation. External framework was reconstructed with bilateral conchal bowl cartilage and rib osteocartilagenous grafts. Patient was pleased with the aesthetics and had safe decannulation with the ability to breathe through the nose and airway.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-22T09:09:31Z
      DOI: 10.1177/10556656211012859
       
  • Changes in the Transverse Dimension of the Maxillary Arch of 5-Year-Olds
           Born With UCLP Since the Introduction of Nationwide Guidance

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      Authors: Charlotte Molyneaux, Martyn Sherriff, Yvonne Wren, Anthony Ireland, Jonathan Sandy
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To determine whether the transverse dimensions of the maxillary arch of 5-year-old children with unilateral cleft lip and palate (UCLP) have changed following centralization of cleft services in the United Kingdom.Design:Retrospective cross-sectional study.Setting:Digital analysis of UCLP maxillary dental casts.Participants:All available maxillary dental casts from 5-year-old participants of the Clinical Standards Advisory Group (CSAG, N = 114) and Cleft Care UK (CCUK, N = 175) studies.Interventions:Quantitative measurements of the intercanine width (ICW), intermolar width (IMW), and the distance from the midline to the greater and lesser side canine (GC/LC) and greater side and lesser side second primary molar (GE/LE). Degree measurements of the greater and lesser arch form angles, arch length, anterior palatal depth (APD), and posterior palatal depth were also measured.Main outcome:Differences between the transverse dimensions of the maxillary arch for the CSAG and CCUK cohorts.Results:In 5 (ICW, IMW, LC, LE, and APD) of the 11 measurements, there was a statistically significant difference between the CSAG and CCUK cohorts. In all of these, the CCUK values were greater than CSAG.Conclusions:There have been small but positive improvements for the transverse maxillary dimensions since centralization of the UK cleft service.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-20T09:02:12Z
      DOI: 10.1177/10556656211028511
       
  • Norwegian Orthodontists’ Experience and Challenges With Treatment of
           Patients With Cleft Lip and Palate

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      Authors: Paul K. Saele, Anne-Kristine Nordrehaug Aastrøm, Harald Gjengedal, Elwalid F Nasir, Manal Mustafa
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:Patients born with cleft lip and/or palate (CL/P) have orthodontic treatment challenges due to maxilla deficiency, malocclusions, and dental abnormalities. In Norway, orthodontic treatment is done by centralized CL/P teams. Due to traveling restrictions, this treatment might be done locally in the future. The experience of Norwegian community orthodontists in managing such patients has not been investigated previously.Objective:To assess Norwegian orthodontists’ management of patients with CL/P and need for further education.Material and Methods:All orthodontists in Norway were sent a questionnaire about their experience, challenges, and knowledge and asked about their need of further theoretical education and clinical training in the management of patients with CL/P.Results:Norwegian orthodontists’ standard of knowledge of CL/P treatment is adequate. However, few respondents have treated a high number of cleft patients. Eighty-six percent of the participants believed that treating CL/P patients involves challenges, such as time-consuming treatment and technical difficulties. Increased perceived need for more education was revealed among participants stated unpreparedness during education (4 folds), encountered challenges, and lack of knowledge (almost 3 folds).Conclusions:The study revealed that community orthodontists in Norway lack experience and acknowledged the challenges in treating patients with CL/P. Most of the respondents perceived a need for additional education and clinical training to treat CL/P patients competently. The findings suggested more focus on patients with CL/P management in the curricula and more collaboration between centralized CL/P teams and community orthodontists.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-20T09:01:48Z
      DOI: 10.1177/10556656211028509
       
  • Speech Outcome and Self-Reported Communicative Ability in Young Adults
           Born With Unilateral Cleft Lip and Palate: Comparing Long-Term Results
           After 2 Different Surgical Methods for Palatal Repair

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      Authors: Petra Peterson, Jill Nyberg, Christina Persson, Hans Mark, Anette Lohmander
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To compare speech outcome and self-reported speech and communicative ability (SOK) in young adults treated with one-stage (OS) or two-stage (TS) palatal repair. Furthermore, to compare with normative data on individuals without cleft lip and palate and to study the relationship between patients’ and experts’ judgments.Design:A cross-sectional group comparison study with long-term follow-up. Participants: Patients born with unilateral cleft lip and palate treated at 2 cleft centers; 17 with OS at 14 months and 25 with TS, soft palate repair at 7 months and hard palate repair at 6.2 years. Pharyngeal flap surgery was performed in 53% (OS) and 24% (TS) of patients.Main Outcome Measure(s):Speech characteristics were blindly assessed by speech and language pathologist from audio recordings, SOK at 19 years of age.Results:No group differences were found. Although the occurrence of nasality symptoms was low in both groups, only 60% (OS)/65% (TS) were assessed with competent velopharyngeal function (VPC). Articulation proficiency (percentage of consonants correct [PCC]) was 91%/97%, the /s/-sound specifically 87%/91%. Good intelligibility was found in 91%/87%. Patient opinion was in agreement with norms and significantly associated with intelligibility (rs = 0.436, P < .01), PCC (rs = −0.534, P < .01), and correct /s/ (rs = −0.354, P < .05).Conclusions:No differences in speech outcome were related to operation method. The low prevalence of VPC was not clearly reflected in nasality symptoms. Patient opinion was related to articulation and intelligibility. A higher burden of care in terms of pharyngeal flap surgery was seen after the OS technique.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-15T09:26:25Z
      DOI: 10.1177/10556656211025926
       
  • Early Communication Behaviors in Infants With Cleft Palate With and
           Without Robin Sequence: A Preliminary Study

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      Authors: Stephanie van Eeden, Yvonne Wren, Cristina McKean, Helen Stringer
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To investigate the early communication behaviors in infants with nonsyndromic isolated cleft palate (iCP) and Robin sequence (RS).Design:Group comparison using parent report.Participants:There were 106 participants included in this study. Two groups were selected from the UK Cleft Collective resource. Parents had completed the Language ENvironment Analysis Developmental Snapshot questionnaire when their child turned 13 months. There were 78 participants in the iCP group and 28 in the RS group.Main Outcome Measure(s):Total number of communication behaviors reported on the questionnaire. Subdomains for expressive and receptive language and social communication behaviors were also analyzed.Results:There were no statistically significant group differences. Parents of infants with RS reported fewer later communication behaviors compared to the iCP group. Infants in both groups had fewer communication behaviors compared to the normative sample. Across the whole sample, post hoc analysis revealed a significant correlation between severity of the cleft and social communication behaviors and expressive but not receptive language. Infants with a cleft of the hard and soft palate were more likely to be in the RS group (odds ratio: 7.04 [95% CI: 1.55-32.04]; P = .01).Conclusions:Both groups reported similar levels of early communication. Some divergence of more complex language skills was seen, although there were no significant group differences. A relationship with the diagnosis of a cleft of the hard or soft palate with expressive language behaviors was found. Further study into the impact of cleft severity on early speech development and the relationship with later language skills is needed along with longitudinal follow-up of this population.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-14T09:10:52Z
      DOI: 10.1177/10556656211031877
       
  • Prevalence of Dental Anomalies in Patients With Unilateral Cleft Lip and
           Alveolus Treated With Gingivoperiosteoplasty

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      Authors: Travis L. Gibson, Barry H. Grayson, Court B. Cutting, Pradip R. Shetye
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To compare the prevalence of dental malformations and agenesis in patients who received or did not receive gingivoperiosteoplasty (GPP).Design:Retrospective cohort study.Patients:Review of patients born January 1, 2000, to December 31, 2007, with unilateral cleft lip and alveolus, with or without clefting of the secondary palate, who received GPP and/or secondary alveolar bone grafting (ABG). Patients were included if they had clinical images and dental radiographs available at ages 5 to 9 and 10 to 12 years. Ninety-four patients met the inclusion criteria; 46 treated with GPP, and 48 who did not receive GPP.Outcome Measures:Records were assessed for supernumerary, missing, and malformed teeth by a blinded examiner, and prevalence compared between groups using χ2 tests.Results:Cleft side lateral incisors were absent in 54% of GPP patients, compared to 50% in the no-GPP group. Two patients in the GPP group and 1 in the no-GPP group had supernumerary lateral incisors. Most lateral incisors were undersized or peg shaped in both the no-GPP (83.3%) and GPP (71.4%) groups. In the GPP group, 5 (10.9%) patients exhibited central incisor agenesis, and 3 had significant hypoplasia. In the no-GPP group, 4 (8.3%) patients exhibited central incisor agenesis, and 5 (10.5%) significant hypoplasia. These differences were not statistically significant.Conclusions:Gingivoperiosteoplasty was not associated with increased prevalence of dental malformation or agenesis. When performed appropriately, GPP is a safe treatment technique that does not increase the risk of dental anomalies.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-14T09:09:52Z
      DOI: 10.1177/10556656211031124
       
  • Oral Colonization and Virulence Factors of Candida spp. in Babies With
           Cleft Palate

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      Authors: Patrícia Tolentino da Rosa de Souza, Nilza Cristina Valor Gonçalves-Wilhelmsen, Rosimeire Takaki Rosa, Clarice de Fatima Kamaroski Noan Correia, Thais Munhoz Pereira, Aldini Beuting Pereira Kitahara, Sérgio Aparecido Ignácio, Luciana Reis Azevedo-Alanis, Edvaldo Antônio Ribeiro Rosa
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To examine oral colonization and virulence factors of Candida spp. in patients aged from 0 to 18 months with cleft palate (CP).Materials and Methods:Sixty babies were allocated into 3 groups: CP, CP with orthodontic plate (CPwP), and control group (Ctrl) without CP. Information on feeding habits, hygiene, and history of candidosis was collected. The presence of Candida spp. was investigated in samples of saliva. Fungal hydrophobicity, protease, esterase, phospholipase, and hemolysin were evaluated in a semiquantitative manner.Results:Positive oral isolations of Candida spp. were detected in CP (89.5%), CPwP (100%), and Ctrl (44%) groups. Candidosis was more reported in the cleft groups than in the Ctrl group (P ≤ .023). There was a higher prevalence of Candida albicans, followed by Candida krusei, Candida tropicalis, and Candida parapsilosis in all groups. There was no uniformity of expression of virulence factors, either among different species or among different groups.Conclusion: Candida spp. colonization occurred in all groups, being superior in CPwP group. Candidosis episodes were more reported in patients from CPwP than in other groups, although candidosis was also registered in other groups. Candida albicans was the predominant species and virulence factors did not exhibit any pattern for species or groups of patients.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-14T09:09:24Z
      DOI: 10.1177/10556656211030437
       
  • Greater Palatal Cleft Width Predicts an Increased Risk for Unfavorable
           Outcomes in Cleft Palate Repair

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      Authors: Åsa C. Okhiria, Fatemeh Jabbari, Malin M. Hakelius, Monica M. Blom Johansson, Daniel J. Nowinski
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To investigate the impact of cleft width and cleft type on the need for secondary surgery and velopharyngeal competence from a longitudinal perspective.Design:Retrospective, longitudinal study.Setting:A single multidisciplinary craniofacial team at a university hospital.Patients:Consecutive patients with unilateral or bilateral cleft lip and palate and cleft palate only (n = 313) born from 1984 to 2002, treated with 2-stage palatal surgery, were reviewed. A total of 213 patients were included.Main Outcome Measures:The impact of initial cleft width and cleft type on secondary surgery. Assessment of hypernasality, audible nasal emission, and glottal articulation from routine follow-ups from 3 to 16 years of age. The assessments were compared with reassessments of 10% of the recordings.Results:Cleft width, but not cleft type, predicted the need for secondary surgery, either due to palatal dehiscence or velopharyngeal insufficiency. The distribution of cleft width between the scale steps on a 4-point scale for hypernasality and audible nasal emission differed significantly at 5 years of age but not at any other age. Presence of glottal articulation differed significantly at 3 and 5 years of age. No differences between cleft types were seen at any age for any speech variable.Conclusions:Cleft width emerged as a predictor of the need for secondary surgery as well as more deviance in speech variables related to velopharyngeal competence during the preschool years. Cleft type was not related to the need for secondary surgery nor speech outcome at any age.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-14T09:05:44Z
      DOI: 10.1177/10556656211029537
       
  • Hearing Outcomes in Stickler Syndrome: Variation Due to COL2A1 and COL11A1

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      Authors: Fadlullah Bath, Dan Swanson, Hanan Zavala, Siva Chinnadurai, Brianne B. Roby
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:Stickler syndrome (SS) is a heterogeneous inherited connective tissue disorder, often due to a mutation in COL2A1 or COL11A1. Mutations in these genes cause collagen abnormalities affecting ocular, auditory, orofacial, and skeletal systems, including hearing loss, micrognathia, and cleft palate. Understanding the variability of hearing phenotypes based on genetic mutation has a significant impact on treatment and long-term care.Design:A retrospective chart review of pediatric patients with a confirmed diagnosis of SS between January 2003 and December 2018 at a tertiary pediatric hospital was performed. Patients were excluded if they did not have genetic evaluation, craniofacial/ear, nose, and throat evaluation, and/or audiologic testing. Charts were reviewed for the following information: age, race, sex, SS diagnosis, genetic variant of SS, and audiological testing data.Results:There were 29 confirmed patients with SS who met criteria, 16 with type I (COL2A1) and 13 with type II (COL11A1). Of the 13 patients with type II, 12 (92%) demonstrated hearing loss, ranging in severity from mild to severe. In type I, 25% of patients had mild or resolved hearing loss.Conclusion:Results suggest that patients with type II SS are more likely to have congenital hearing loss than type I. Data also suggest that the COL11A1 mutation shows consistently more severe hearing loss than the COL2A1 mutation.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-09T08:46:25Z
      DOI: 10.1177/10556656211029519
       
  • Familial Pfeiffer Syndrome: Variable Manifestations and Role of
           Multidisciplinary Team Care

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      Authors: Sarut Chaisrisawadisuk, Mark H Moore
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Pfeiffer syndrome is one of the autosomal dominant craniofacial syndromes. Classical clinical manifestations are coronal suture synostosis causing brachycephaly, midface retrusion, airway compromise, broad thumbs, and toes. Pfeiffer syndrome type I (classic type) is associated with FGFR1 mutation. However, wide range of clinical manifestations, with and without craniosynostosis, have been reported. Here, we present a family of Pfeiffer syndrome across 3 generations with identical FGFR1: c.755C>G (p.Pro252Arg) mutation. Where the members of the youngest generation have no cranial involvement. Lastly, we propose a guideline management for familial Pfeiffer syndrome management.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-09T08:39:13Z
      DOI: 10.1177/10556656211028505
       
  • Assessing Adherence to Audiologic Parameters of Care for Children With
           Cleft Palate: A Quality Improvement Initiative

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      Authors: Ursula M. Findlen, Jonathan Grischkan, Sandra Alston, Lauren Durinka, Adriane Baylis
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate and increase adherence to an evidence-based audiologic management protocol for children with cleft palate.Design:Prospective, multidisciplinary quality improvement initiative.Setting:Tertiary pediatric hospital.Patients, Participants:Children with cleft palate (with or without cleft lip) between the ages of 0 and 5 years (n = 205).Interventions:A multidisciplinary team identified key drivers for nonadherence to recommended audiological follow-up and implemented interventions to improve adherence. Key drivers included provider practices and preferences, clinic logistics and flow, and patient/family awareness and education. Several interventions were implemented between 2016 and 2020, including developing an evidence-based audiologic protocol, maximizing access to audiologic clinic visits across multiple departments, cleft team education, and improved team communication.Main Outcome Measure(s):Completion of recommended audiologic assessment at 5 separate care milestones.Results:After implementation of interventions between 2016 and 2020, adherence to recommended audiologic follow-up increased from 59% to 84%. Analysis of individual care milestones revealed that increased access to audiologic testing during team clinics resulted in the largest increase in adherence to recommended follow-up. Additionally, cause-effect analysis revealed that nonadherence due to provider-related causes decreased over the project period to a greater extent than patient/family-related causes.Conclusions:Implementation of an evidence-based audiologic care protocol and improvements in access to early hearing care are feasible in a high-volume multidisciplinary cleft clinic. Adherence to recommended audiologic management can be improved by establishing strategies to improve access to care, team member and family education, and enhanced team communication.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-08T09:22:58Z
      DOI: 10.1177/10556656211029526
       
  • Final Posttreatment Occlusion in Patients With Unilateral Cleft Lip and
           Palate

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      Authors: Christine B. Staudt, Julia Bollhalder, Martina Eichenberger, Giorgio La Scala, Georges Herzog, Daniel B. Wiedemeier, Gregory S. Antonarakis
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate final posttreatment occlusion in patients with complete unilateral cleft lip and palate (cUCLP) by comparing (1) 3 treatment centers, (2) males and females, (3) cleft and noncleft sides, (4) right- and left-sided clefts, and (5) orthodontic treatment with/without orthognathic surgery (OS).Design:Retrospective cohort study.Patients:Blinded posttreatment dental casts of 56 patients (19.4 ± 1.4 years) with cUCLP from 3 centers in Switzerland.Main Outcome Measure:Occlusal assessment using the modified Huddart/Bodenham (MHB) index.Results:Our sample comprised 35 males and 21 females, 46 with left- and 10 with right-sided clefts, of which 32 had undergone OS. The final posttreatment occlusion showed a median MHB score of 0 (interquartile range: −1.0 to 2.0) in the total sample and did not seem to depend on treatment center, sex, or OS. The MHB scores for the anterior buccal and the buccal segments were more negative on the cleft than on the noncleft side (P = .002 and P = .006, respectively). When the cleft was on the left side, the MHB score tended to be more positive in the labial (P = .046) and anterior buccal segments (P = .034).Conclusions:This study shows a very satisfactory final posttreatment occlusion in patients with cUCLP. The more constricted buccal occlusion on the cleft side emphasizes the attention that should be given in correcting the more medially positioned lesser maxillary segment. The influence of cleft-sidedness should be analyzed further on a sample including more patients with right-sided clefts.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-08T09:21:57Z
      DOI: 10.1177/10556656211028506
       
  • Factors Associated With Psychological Adjustment in Adults With Cleft Lip
           and/or Palate: Findings From a National Survey in the United Kingdom

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      Authors: Bruna Costa, Kenny Ardouin, Nicola Marie Stock
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:Research has identified adults born with cleft lip and/or palate (CL/P) to be at risk of poorer psychological outcomes compared to the general population. This study investigated factors that may contribute to positive and negative adjustment in adults born with CL/P.Design:A survey was designed and distributed by the Cleft Lip and Palate Association in collaboration with the Centre for Appearance Research CAR at the University of the West of England (UWE). There were 207 eligible responses (95% completed online) received between July and October 2018. Dependent variables included the Body Esteem Scale for Adolescents and Adults, Harter’s Self Perception Profile for Adults (Global Self-Worth, Social Competence, and Intimacy subscales), the Fear of Negative Appearance Evaluation Scale, and the Revised Adult Attachment Scale. Independent variables were the Revised Life Orientation Test, biodemographic data, and self-reported single-item questions.Results:Factors associated with positive adjustment included reports of a happy childhood, talking about CL/P with family, close friendships, comfort in public spaces, satisfaction with appearance, and a positive life orientation. Psychological distress was associated with a desire for further surgery to improve appearance and/or function.Conclusions:Several factors were identified that may influence psychological adjustment in adults with CL/P. Throughout childhood, family-centered practice to support family cohesion and an open dialogue about CL/P is indicated, as is support for young people to develop social confidence. For adults returning to the cleft service, treatment options for appearance and/or functional concerns should be explored, with access to psychological support when indicated. Interventions to increase optimism, resilience, and self-acceptance may also be warranted throughout the life span.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-08T09:21:28Z
      DOI: 10.1177/10556656211028494
       
  • The Prevalence and Morphology of Supernumerary Teeth in Children With
           Nonsyndromic Cleft Lip and Palate

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      Authors: Agnieszka Lasota, Weronika Siebieszuk, Przemysław Pastuszak, Adrianna Mostowska
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:Cleft lip with or without cleft palate (CL/P) is congenital deformity associated with hyperdontia.Objective:To determine the prevalence and characteristics of supernumerary teeth in patients with CL/P.Design:Retrospective descriptive and correlation clinical study.Patients:One hundred thirteen children with cleft (age ranged 9.3-19.2; 67 males and 46 females) treated in Clinic of Congenital Facial Deformities Medical University of Lublin were included in the study.Methods:Records evaluation was conducted regarding age, gender, cleft type (Q36, Q37—International Classification of Diseases 10th revision), cleft side, and incidence of supernumeraries. In all supernumerary teeth, size, shape, and developmental degree were analyzed and correlation between the incidence of hyperdontia with different variables was checked. Correlations were detected using chi-square and the Yates correction.Results:The majority of the examined group were males—59.29% with Q37 (67.26%) and the cleft on the left side (62.83%). Hyperdontia was noted in 26.55%. Only upper lateral incisors were affected. They usually had atypical shape (56.67%), reduced size (83.33%), and delayed development (56.67%).Conclusions:The prevalence of supernumerary permanent teeth in patients with cleft was higher than in the general population. Anomaly was more frequent in male patients and occurred mainly on the cleft side. The severity of the cleft did not influenced the frequency of supernumerary teeth, their shape, size, and developmental degree. Supernumerary teeth were characterized by reduced crown size, abnormal structure, incorrect inclination, and delayed development phase.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-05T09:25:17Z
      DOI: 10.1177/10556656211027750
       
  • A New Frame for Orienting Infants With Cleft Lip and Palate During
           3-Dimensional Facial Scanning

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      Authors: KH Alisha, Puneet Batra, Sreevatsan Raghavan, Karan Sharma, Aditya Talwar
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:Nonradiographic 3-D assessment of soft tissue facial structures is an ideal tool to measure 3-D facial change and smartphone apps like Bellus 3D have brought these technologies to our doorsteps. Although this app works well for adults, it doesn’t do so well with infants as their stabilization is difficult thus proper alignment becomes problematic often leading to distortion. This seriously hampers the repeatability and reliability of the whole process.Material and Method:A stainless steel rod of 150-cm length is curved into a semicircle of a radius of 42.5 cm. Bases are fabricated at both the ends to firmly hold the stand on the table. A Teflon ring of one inch is made for adjusting the diameter of the camera holder. A camera holder is then attached to the Teflon ring. The mobile holder mounted is free to move within the arc. A 3-D facial scan of a single patient was carried out with the camera mounted on the frame and once without the frame. The same was compared using side-by-side comparison and superimposition of the .STL files from the frontal, profile, and worm’s eye view.Result and Conclusion:It was found that considerable distortion was noted around the nasal and paranasal areas when the scan was performed without the frame as compared to the one that was taken with the frame. This technique avoided distortion and the difficulty in turning the baby’s head to record the lateral portion of the face.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-05T09:24:46Z
      DOI: 10.1177/10556656211028512
       
  • SHORT Syndrome: Systematic Appraisal of the Medical and Dental Phenotype

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      Authors: Suhaym Mubeen, Clara Gibson, Raiyan Mubeen, Sahar Mansour, Robert D. Evans
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:SHORT syndrome is a rare autosomal dominant condition described by its acronym of short stature, hyperextensibility of joints and/or inguinal hernia, ocular depression, Rieger abnormality, and teething delay. Individuals have a distinct progeroid craniofacial appearance with a triangular face, frontal bossing, hypoplastic or thin alae nasi, large low-set ears, and mandibular retrognathia.Objectives:To systematically appraise the literature and update the clinical phenotype with emphasis on the dental condition.Design:A systematic literature search was carried out to update the clinical phenotype, identifying reports of individuals with SHORT syndrome published after August 2015. The same search strategy but not limited to publication date was carried out to identify reports of the dental phenotype. Two independent reviewers screened 1937 articles with 55 articles identified for full-text review.Results:Nineteen individuals from 11 families were identified. Facial dysmorphism including ocular depression, triangular shaped face, frontal bossing, large low-set ears, and micrognathia were the most consistent features followed by lipodystrophy, insulin resistance, and intrauterine growth restriction. Teething delay, microdontia, hypodontia, and enamel hypoplasia have all been reported.Conclusion:Features that comprise the SHORT acronym do not accurately or completely describe the clinical phenotype. The craniofacial appearance is one of the most consistent features. Lipodystrophy and insulin resistance may also be considered cardinal features. After teething delay, enamel hypoplasia and microdontia are the most common dental manifestations. We present recommendations for the dental and orthodontic/orthognathic management of individuals with SHORT syndrome.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-02T09:03:55Z
      DOI: 10.1177/10556656211026859
       
  • A Retrospective Longitudinal Treatment Review of Multidisciplinary
           Interventions in Nonsyndromic Robin Sequence With Cleft Palate

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      Authors: Pinelopi K. Palaska, Gregory S. Antonarakis, Sunjay Suri
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To document and analyze the overall longitudinal institutional treatment experience of children with nonsyndromic Robin sequence (RS) from infancy to early adulthood.Design:Retrospective longitudinal treatment review.Setting:A tertiary-care, referral, teaching hospital.Patients:Children with nonsyndromic RS and cleft palate (N = 117) born between December, 1985, and January, 2012.Interventions:Data regarding airway management, nutritional management, audiological interventions, orthodontic treatment, and surgical interventions were documented and analyzed in different growth/developmental stages. Comparative data from other international centers were collected from the literature.Results:Airway management during infancy involved prone positioning (92%), nasopharyngeal airway (6%), tracheostomy (2%), and mandibular distraction osteogenesis (1%). Feeding with nasogastric, gastrostomy, and/or gastrojejunostomy tubes was used in 44%, Haberman feeders in 53%, and Mead Johnson feeders in 3%. Gastroesophageal reflux disease was documented in 6% of the sample. During childhood and early adolescent years, pharyngeal flap surgery was carried out in 22% of the children, while 11% had secondary palatal surgery. Audiological management included the use of tympanostomy tubes in 62%, with several children needing multiple tube replacements. At least 18% were diagnosed with obstructive sleep apnea. Adenoidectomy or adenotonsillectomy was undertaken in 4%. Analysis of data pertaining to middle childhood and adolescent years showed that orthodontic treatment was conducted for most children for crowding, tooth agenesis, and skeletal and/or dental dysplasia. Orthognathic surgery frequency (
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-02T09:03:13Z
      DOI: 10.1177/10556656211026477
       
  • Tessier 30 Facial Clefts—A Literature Review of 72 Cases (1996-2020),
           Suggested Treatment Protocol, Outcome Measures, Minimum Dataset for Future
           Case Reports, and Registries

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      Authors: Ruben van der Valk, Stephen Magill, Annie Pellatt, Nazanin Ahmadi-Lari, Simon P. Hall, Alistair R. M Cobb, Tom W. M. Walker
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:Tessier 30 facial cleft is a rare anomaly presenting in the soft and hard tissues over the central lower face. Owing to the rarity of cases and difficulty of treatment, there is no universally accepted surgical management strategy. The last comprehensive literature review of Tessier 30 clefts was in 1996. This report aims to update the literature to inform decision-making on treating Tessier 30 cases.Methodology:A literature search was performed. PubMed, SCOPUS, and OVID databases were searched. A total of 72 cases in 51 articles were analyzed, looking at demographics, extent of cleft, parent health, family history, procedures, follow-up, existence of other anomalies, and stages of repair.Results:Surgeons are increasingly choosing to repair Tessier 30 defects in one rather than multiple stages. Of the 72 cases studied, only 31 had documented the completed repair of the cleft. All completed soft tissue only defects were repaired in 1 stage of repair (n = 11). Where both soft tissue and mandible was involved (n = 20), 55% (n = 11) had undergone 1-stage repair to address the Tessier 30 cleft.Discussion:We argue that a single-stage approach is preferable to multistage. Primary mucogingivoperiosteoplasty should be undertaken in children at the time of management of the soft tissue cleft. The timing of this procedure should be in the latter half of the first year of life, as this is when mandibular symphyseal fusion normally occurs. We have suggested a treatment protocol and we hope that future case reports use our minimum data set.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-07-01T09:00:17Z
      DOI: 10.1177/10556656211019237
       
  • Care Barriers for Patients With Nonsyndromic Orofacial Clefts in Saudi
           Arabia: A Cross-Sectional Study

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      Authors: Hanaa Allaf, Narmin Helal, Osama Basri, Ahmad AlShadwi, Heba Sabbagh
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate the barriers faced by patients with nonsyndromic orofacial clefts (NSOFC) throughout their treatment course in Saudi Arabia.Design:A cross-sectional study.Setting:Eleven different governmental health care centers across Saudi Arabia. Patients: Records of pediatric patients with NSOFC.Interventions:A questionnaire with multiple validation stages was designed to assess the barriers in care of these patients through telephonic interviews with the parents or guardians of patients with NSOFC.Main Outcome Measures:We identified 3 care-barrier–related factors: (1) geographic accessibility, (2) appointment availability and accessibility, and (3) scheduling-related barriers.Results:Overall, 240 participants of both sexes, with orofacial cleft of various types and with various demographic characteristics (residence, family monthly income, and caregiver level of education) were included. The highest mean score of care barriers was reported for scheduling-related barriers. Overall, 186 individuals reported sometimes/often not receiving the required medical care for the following reasons: scheduling difficulties (89%; 37.1%), prolonged waiting room time (40%; 16.7%), and transportation difficulties (36%; 15.0%). A linear regression showed that parents cited late appointments as the main reason for patients with NSOFC not receiving adequate medical care. Care-barrier factors were significantly related to gender (P = .035), patient age (P < .001), place of residency (P < .001), and caregiver’s level of education (P = .015).Conclusions:Gaps in the health care system directly related to common care barriers need to be addressed to ensure adequate care for patients with NSOFC.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-30T09:31:02Z
      DOI: 10.1177/10556656211026536
       
  • Non-Oral Compensatory Misarticulations Revisited

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      Authors: Mary A. Hardin-Jones, Kathy L. Chapman
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Non-oral compensatory misarticulation patterns are often associated with the speech of children with cleft palate. Despite their saliency, the etiology, frequency, and treatment of these misarticulations have not been studied extensively. The purpose of this commentary is to review what we know about these atypical patterns of articulation and address clinical assumptions regarding their etiology and treatment.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-29T09:38:19Z
      DOI: 10.1177/10556656211026488
       
  • Expanding Support Services for Adults Born With Cleft Lip and/or Palate in
           the United Kingdom: An Exploratory Evaluation of the Cleft Lip and Palate
           Association Adult Services Programme

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      Authors: Kenny Ardouin, Nicky Davis, Nicola Marie Stock
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:The largest group of people living with repaired cleft lip and/or palate (CL/P) are adults. Previous research has identified unmet treatment and psychosocial needs, yet few interventions exist. This article outlines 3 interventions developed by the Cleft Lip and Palate Association as part of its 3-year community-based Adult Services Programme; an Adults Conference, a series of panel discussions (“Cleft Talk”) streamed in podcast/video format, and a Leaver’s Pack of resources for adults wishing to return to cleft care.Methods:Feedback from attendees of the Adults Conferences (2018-2019) was collected using specifically developed evaluation forms. Streaming metrics and social media interactions were extracted for Cleft Talk panel discussions (2019-2020). The Leaver’s Pack was piloted in 2020, using an online evaluation form. Specialist health professionals were invited to provide feedback or participate in a one-to-one interview regarding their perceived impact of the program.Results:All 3 interventions across the different modalities received support from participating adults, demonstrating potential to meet adults’ needs across the life span. Health professionals also offered support for the program, viewing the interventions as a valuable adjunct to formal medical CL/P services.Conclusions:This exploratory evaluation indicates that peer- and community-led interventions, in combination with ongoing access to specialist medical care, can have a range of positive impacts for adults with CL/P. There is scope for similar initiatives to be developed internationally and for individuals with other craniofacial conditions. Not-for-profit organizations are encouraged to routinely evaluate their interventions to create a stronger evidence base for their valuable work.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-29T09:37:30Z
      DOI: 10.1177/10556656211025415
       
  • Nasalance Scores for Normal Speakers of American English Obtained by the
           Nasometer II Using the MacKay-Kummer SNAP-R Test

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      Authors: Firas Alfwaress, Ann W. Kummer, Barbara Weinrich
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To establish nasalance score norms for adolescent and young adult native speakers of American English and also determine age-group and gender differences using the Simplified Nasometric Assessment Procedures (SNAP) Test-R and Nasometer II.Design:Prospective study using a randomly selected sample of participants.Setting:Greater Cincinnati area and Miami University of Ohio.Participants:Participants had a history of normal speech and language development and no history of speech therapy. Participants in the adolescent group were recruited from schools in West Clermont and Hamilton County, whereas the young adults were recruited from Miami University of Ohio. The participants of both groups were residents of Cincinnati, Ohio or Oxford, Ohio and spoke midland American English dialect.Outcome Measures:Mean nasalance scores for the SNAP Test-R.Results:Normative nasalance scores were obtained for the Syllable Repetition/Prolonged Sounds, Picture-Cued, and Paragraph subtests. Results showed statistically significant nasalance score differences between adolescents and young adults in the Syllable Repetition, Picture-Cued, and Paragraph subtests, and between males and females in the Syllable Repetition and the Sound-Prolonged subtests. A significant univariate effect was found for the syllables and sentences containing nasal consonants and high vowels compared to syllables and sentences containing oral consonants and low vowels. Across all the SNAP Test-R subtests, the females’ nasalance scores were higher than the males. A significant univariate effect was also found across nasal syllables, and high vowels such that the females’ nasalance scores were higher than the males. Tables of normative data are provided that may be useful for clinical purposes.Conclusion:Norms obtained demonstrated nasalance score differences according to age and gender, particularly in the Syllable Repetition/Prolonged Sound subtest. These differences were discussed in light of potential reasons for their existence and implications for understanding velopharyngeal function. In addition, nasalance scores are affected by the vowel type and place of articulation of the consonant. These facts should be considered when nasometry is used clinically and for research purposes.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-29T09:36:57Z
      DOI: 10.1177/10556656211025406
       
  • Effectiveness of Presurgical Nasoalveolar Molding Appliance in Infants
           With Complete Unilateral Cleft Lip and Palate

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      Authors: Thao Thi Nhu Dinh, Dau Van Nguyen, Vu Hoa Anh Dien, Tham Khac Dong
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To assess the effectiveness of the presurgical nasoalveolar molding appliance among infants with unilateral cleft lip and palate.Methods:In this prospective study, 95 pairs of casts of infants with unilateral cleft lip and palate treated by presurgical nasoalveolar molding were selected at the Children’s Hospital 1 at Ho Chi Minh City, Vietnam. The average time of treatment was 3 months. All casts were scanned and measured using 3-dimensional technology before and after treatment. Paired t tests were applied for comparisons.Results:There was a statistically significant increase in the nostril height in cleft side (P < .001), decrease in the nostril width and columella angle (P < .001), and decrease in cleft width and midline deviation (P < .001) after treatment with presurgical nasoalveolar molding appliance.Conclusions:Nasoalveolar molding appliance is effective in improving the morphology of nostril and maxillary alveolar. Understanding this helps orthodontists and surgeons in treatment outcome expectations.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-24T09:04:58Z
      DOI: 10.1177/10556656211026493
       
  • Improvement in Total and Face-to-Face Provider Time in a Multidisciplinary
           Craniofacial Team Clinic: An Interventional Study

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      Authors: Alexis C. Wood, C. Alejandra Garcia de Mitchell, Ruchi Kaushik
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Identify factors contributing to time a family spends in a Multidisciplinary Craniofacial Team Clinic (MDCT) and implement an intervention to reduce this time.Design:Interventional: a restructuring of clinics to serve those patients requiring fewer provider encounters separately.Setting:An American Cleft Palate-Craniofacial Association-accredited MDCT in an academic children’s hospital.Patients/Participants:One hundred sixty-seven patients with craniofacial diagnoses.Interventions:Time data were tabulated over ∼2 years. Following 9 months of data collection, patients requiring fewer provider encounters were scheduled to a separate clinic serving children with craniosynostosis, and data were collected in the same fashion for another 14 months.Main Outcome Measures:Principal outcome measures included total visit time and proportion of the visit spent without a provider in the room before and after clinic restructuring.Results:The average time spent by family in a clinic session was 161.53 minutes, of which 64.3% was spent without a provider in the room. Prior to clinic restructuring, a greater number of provider encounters was inversely associated with percentage of time spent without a provider (P < .001). Upon identifying this predictor, scheduling patients who needed fewer provider encounters to a Craniosynostosis Clinic session resulted in reduction in absolute and percentage of time spent without a provider (P < .001).Conclusions:The number of provider encounters is a significant predictor of the proportion of a clinic visit spent without a provider. Clinic restructuring to remove patient visits that comprise fewer provider encounters resulted in a greater percentage of time spent with a provider in an MDCT.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-24T09:04:18Z
      DOI: 10.1177/10556656211021705
       
  • Interdisciplinary Approach for the Treatment of Complex Bilateral Cleft
           Lip and Palate With Missing Premaxilla

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      Authors: Yong Jong Park, Christopher Derderian, Michael Oppedisano
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Complex craniofacial deformities such as complete bilateral cleft lip and palate require interdisciplinary approach for proper diagnosis and treatment. A severe skeletal discrepancy caused by bilateral cleft lip and palate and missing premaxilla was successfully managed with orthodontic preparation and distraction osteogenesis. Conventional prosthodontic treatment combined with orthodontic preparation was proven to be a viable option to manage multiple missing teeth in cleft lip and palate.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-23T10:54:30Z
      DOI: 10.1177/10556656211023241
       
  • Systematic Review of Clinical Practice Guidelines for Oral Health in
           Children With Cleft Lip and Palate

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      Authors: Ankita Saikia, M.S. Muthu, Omolola O. Orenuga, Peter Mossey, Lahcen Ousehal, Si Yan, Marina Campodonico, Rachael England, Sean Taylor, Pamela Sheeran
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Clinical practice guidelines (CPGs) exist to present recommendations and policies aimed at optimizing the oral health of children and adolescents born with cleft lip and/or palate. The aim of this review is to identify and assess the scope, quality, adequacy, and consistency of CPGs related to oral health in children and adolescents with clefts, along with reporting any differences and shortcomings.Methods:A systematic review of the literature of CPGs following Preferred Reporting Items for Systematic Reviews guidelines was conducted. Assessment of selected CPGs was performed using the Appraisal of Guidelines for Research & Evaluation II methodological quality instrument.Results:Only 7 CPGs fulfilled the criteria. Of these, 4 were from the American Cleft Palate-Craniofacial Association, and 1 each from the American Academy of Pediatrics, the Academy of Breastfeeding Medicine, and the American Academy of Pediatric Dentistry. The lowest overall mean scores were in the domain “Rigor of Development” (mean 29.58%, SD 17.11), revealing lower quality in methodology of the guideline. The domain “Clarity of Presentation” (mean 73.80%, SD 7.87) revealed the best score.Conclusions:Our review results reveal a lack of integrated high-quality CPGs that can be used as universal guidelines by health workers in a range of disciplines for improving oral health in children and adolescents with cleft problems.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-23T10:47:55Z
      DOI: 10.1177/10556656211025189
       
  • Does the Type of MRI Sequence Influence Perceived Quality and Measurement
           Consistency in Investigations of the Anatomy of the Velopharynx'

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      Authors: Jamie L. Perry, Abigail E. Haenssler, Katelyn J. Kotlarek, Joshua Y. Chen, Xiangming Fang, Yifan Guo, Kazlin Mason, Michael Webb
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Purpose:The purpose of this study was to evaluate perceived image quality, confidence in identifying key velopharyngeal landmarks, and reliability of making velopharyngeal measures between 3-dimensional (3-D) and 2-D magnetic resonance imaging (MRI) methods and between T1-, T2-, and proton density (PD)-weighted sequences.Methods:Twelve healthy participants completed an MRI study. Three raters assessed overall image quality and their ability to identify key anatomic features within the images. A single rater evaluated the reliability of making measures between imaging methods and sequence types to determine if image type (2-D and 3-D) or image sequence (T1, T2, PD weighted) resulted in different values for key velopharyngeal landmarks.Results:An analysis of variance test revealed image quality was rated significantly different based on the scan type (P < .001) and the sequence used (P = .015). Image quality was rated higher among 2-D MR images compared to 3-D, and higher among T2 sequences compared to T1- and PD-weighted imaging methods. In contrast, raters favored 3-D sequences over 2-D sequences for identifying velopharyngeal landmarks. Measures of reliability revealed scan type significantly impacted 2 of the 6 variables but to a minimal degree; however, sequence type had no impact on measures of reliability across all variables.Conclusion:Results of the study suggest the scan type and sequence used are factors that likely do not impact the reliability of measures. Based on image quality, the recommended technique for velopharyngeal imaging would be using a 2-D T2-weighted technique. However, based on the ability to identify key landmarks, a 3-D T1- or PD-weighted technique was favored.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-22T09:26:02Z
      DOI: 10.1177/10556656211025191
       
  • Commentary of Early Weight Gain in Infants With Cleft Lip and Palate
           Treated With and Without Nasoalveolar Molding: A Retrospective Study

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      Authors: Renato da Silva Freitas, Maria Cecília Closs Ono
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.

      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-22T09:19:02Z
      DOI: 10.1177/10556656211022912
       
  • Modified Z-Palatoplasty for Correction of Acquired Nasopharyngeal Stenosis
           Following Palatal Surgery: A Case Series

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      Authors: Mohamed Eesa, Ehsan Hendawy, Mohammad Waheed El-Anwar
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:To evaluate the efficacy of a novel surgical technique in management of nasopharyngeal stenosis (NPS), describing its steps and results. Study Design: Prospective clinical trial.Setting:This study was conducted at the Otolaryngology, Head and Neck Surgery Department, Zagazig University.Methods:This prospective study was conducted on patients with snoring ± obstructive sleep apnea due to acquired postsurgical NPS of grade Ι and ΙΙ. New surgical repair was employed on the patients and the pre and postoperative results were statistically compared.Results:The grade of NPS improved significantly postoperatively (P = .00136) throughout a follow-up of 1 year. Postoperatively, there was statistically significant improvement of apnea hypopnea index (P = .0005), Visual Analog Scale (VAS) of nasal obstruction (P < .0001) and VAS of snoring (P < .0001). Dysphagia showed early worsening, but it improved completely at 3 months postoperatively.Conclusion:The utilized novel procedure appears effective, low cost, and easily applicable, and it does not require implants, special tools, or suture materials. Furthermore, it gives excellent results, with negligible pain, and rapid recovery without significant complications.Level of Evidence:4.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-22T09:14:53Z
      DOI: 10.1177/10556656211021702
       
  • Evaluation of Maxillary Sinus Characteristics in Patients With Cleft Lip
           and Palate Using Cone Beam Computed Tomography

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      Authors: Maryam Paknahad, Aida Pourzal, Mohammad Mahjoori-Ghasrodashti, Leila Khojastepour
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:The detection of anatomical variation impairments in patients with cleft lip and palate (CLP) is crucial in combined orthodontic and maxillofacial surgical treatments. Therefore, this study aimed at evaluating the anatomical maxillary sinus characteristics, nasal septum deviation (NSD), and mucosal thickening of the maxillary sinus in patients with CLP using cone beam computed tomography (CBCT).Design:The CBCT images were classified into 3 groups of unilateral cleft lip and palate (UCLP; n = 40), bilateral cleft lip and palate (BCLP; n=14), and noncleft (control; n = 54). Subsequently, the maxillary sinus linear dimensions were assessed. Height, width, depth, infundibulum height, and ostium width were assessed as quantitative measures, whereas nasal septum deviation and mucosal thickening were assessed as qualitative measures. One-way analysis of variance and χ2 tests were utilized to identify any significant differences among the groups regarding the aforementioned variables.Results:Significant differences were observed among the groups regarding maxillary sinus height and depth, NSD, and mucosal thickening. Moreover, UCLP and BCLP groups showed higher incidence of NSD and mucosal thickening. However, the size of maxillary sinus height and depth was lower in the UCLP and BCLP groups.Conclusion:The results showed that BCLP and UCLP groups obtained lower maxillary sinus height and depth compared to the control group. On the other hand, incidence of the NSD and mucosal thickening was significantly higher in UCLP and BCLP groups than those in the control group.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-21T09:19:30Z
      DOI: 10.1177/10556656211023239
       
  • Sociodemographics and Quality of Life in Dutch-Speaking Adolescents and
           Adults With and Without a Cleft Lip and/or Palate

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      Authors: Cassandra Alighieri, Evelien D’haeseleer, Kim Bettens, Katrien Bonte, Hubert Vermeersch, Nele Vermeire, Merel Claeys, Daniel Sseremba, George Galiwango, Kristiane Van Lierde
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To investigate the impact of living with a cleft lip and/or cleft palate (CL/P) on sociodemographic variables, quality of life, aesthetics, life satisfaction, and social distress in Dutch-speaking adolescents and adults.Design:Cross-sectional study.Participants:Thirty Dutch-speaking participants with a CL/P with a mean age of 26.93 years (SD = 11.69) and an age- and gender-matched control group of 30 participants (19 men and 11 women) without a CL/P with a mean age of 26.87 years (SD = 11.73).Main Outcome Measures:Self-reported outcomes of sociodemographics, quality of life, aesthetics, life satisfaction, social distress, and impact of cleft on well-being and functioning.Results:No significant differences in educational level, employment, monthly net income, marital status, and having children were found between participants with and without a CL/P. In addition, quality of life, overall aesthetics, life satisfaction, and social distress did not differ between the 2 groups. Among participants with CL/P, there were no gender differences in the influence of their CL/P on daily functioning, well-being, social contacts, family life, applying for a job, work, education, or leisure time.Conclusion:The findings revealed no differences between participants with and without a CL/P with regard to sociodemographics, quality of life, aesthetics, life satisfaction, or social distress. There were no gender differences in the influence of cleft on well-being and functioning. Longitudinal research can help determine possible fluctuations in the impact of living with a CL/P across the life span.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-18T10:09:14Z
      DOI: 10.1177/10556656211024513
       
  • Comparative Assessment of Autogenous Cancellous Bone Graft and
           Bovine-Derived Demineralized Bone Matrix for Secondary Alveolar Bone
           Grafting in Patients With Unilateral Cleft Lip and Palate

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      Authors: Vijay Kumar, Vidya Rattan, Sachin Rai, Satinder Pal Singh, Jai Kumar Mahajan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Comparison between bovine-derived demineralized bone matrix (DMBM) and iliac crest graft over long term for secondary alveolar bone grafting (SABG) in patients with unilateral cleft lip and palate (UCLP) in terms of radiological and clinical outcomes.Design:Prospective, randomized, parallel groups, double-blind, controlled trial.Setting:Unit of Oral and Maxillofacial Surgery, Oral Health Science Centre, Postgraduate Institute of Medical Education & Research, Chandigarh.Participants:Twenty patients with UCLP.Interventions:Patients were allocated into group I (Iliac crest bone graft) and group II (DMBM) for SABG. Outcomes were assessed at 2 weeks, 6 months, and then after mean follow-up period of 63 months.Outcomes Measures:Volumetric analysis of the grafted bone in the alveolar cleft site was done through cone beam computed tomography using Cavalieri principle and modified assessment tool. Clinical assessment was performed in terms of pain, swelling, duration of hospital stay, cost of surgery, alar base symmetry, and donor site morbidity associated with iliac crest harvesting.Results:Volumetric analysis through Cavalieri principle revealed comparable bone uptake at follow-up of 6 months between group I (70%) and group II (69%). Modified assessment tool showed no significant difference between horizontal and vertical bone scores over short- and long-term follow-up. In group II, there was higher cost of surgery, but no donor site morbidity unlike group I.Conclusions:Demineralized bone matrix proved analogous to iliac crest bone graft as per volumetric analysis over shorter period. However, although statistically insignificant, net bone volume achieved was lower than the iliac crest graft at longer follow-up.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-17T09:06:33Z
      DOI: 10.1177/10556656211025197
       
  • Influence of Gender, Dispositional Optimism, and Coping Strategies on
           Appearance-Related Distress Among Swedish Adults With Cleft Lip and Palate
           

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      Authors: Anna Paganini, Martin Persson, Hans Mark
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To investigate the influence of gender, dispositional optimism, and coping strategies on appearance-related distress among individuals with unilateral cleft lip and palate (UCLP).Design:Cross-sectional design with self-report questionnaires analyzed primarily with Spearman correlations (r s) and multivariate regression analyses.Setting:A tertiary cleft center in Sweden.Participants:Eighty individuals with UCLP born 1966 to 1986. The mean age for men (n = 50) and women (n = 30) was 38.8 and 37.4 years, respectively.Main Outcome Measures:The Derriford Appearance Scale 24 measured appearance-related distress, the Life Orientation Test–Revised, short version measured dispositional optimism and pessimism, and the Coping Orientation to Problems Experienced, short version included 14 coping strategies.Results:Women had higher appearance-related distress than men, which was significantly (P < .05) related to self-blame (r s = 0.59), pessimism (r s = 0.59), and low optimism (r s = −0.56). Men’s appearance-related distress was significantly associated with low active coping (r s = 0.35), low use of emotional support (r s = 0.29), denial (r s = 0.39), behavioral disengagement (r s = 0.41), and pessimism (r s = 0.28). The only significant gender interaction reflected greater impact of optimism in reducing appearance-related distress for women (β = −0.06).Conclusions:This study showed that high levels of dispositional optimism decrease appearance-related distress, particularly for women. The coping strategies used differed between men and women, and the results suggest that both gender and psychosocial facto r s need to be considered in regard to appearance-related distress among individuals with UCLP in both clinical and research settings. A possible way to decrease distress is to strengthen positive coping strategies and dispositional optimism.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-17T09:05:11Z
      DOI: 10.1177/10556656211025196
       
  • Intracranial Volume Not Correlated With Severity in Trigonocephaly

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      Authors: Otto D. M. Kronig, Sophia A. J. Kronig, Léon N. A. Van Adrichem
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:Severity of trigonocephaly varies and potentially affects intracranial volume (ICV) and intracranial pressure (ICP). The aim of this study is to measure ICV in trigonocephaly patients and compare it to normative data and correlate ICV with the severity of the skull deformity according to UCSQ (Utrecht Cranial Shape Quantifier).Design:Retrospective study.Setting:Primary craniofacial center.Patients, Participants:Nineteen preoperative patients with nonsyndromic trigonocephaly (age ≤12 months).Intervention:Intracranial volume was measured on preoperative computed tomography (CT) scans by manual segmentation (OsiriX Fondation). Utrecht Cranial Shape Quantifier was used to quantify the severity of the skull deformity. When present, papilledema as sign of elevated ICP was noted.Main Outcome Measures(s):Measured ICV was compared to Lichtenberg normative cranial volume growth curves, and Pearson correlation coefficient was used to correlate UCSQ with the ICV.Results:Mean age at CT scan was 6 months (2-11). Mean measured ICV was 842 mL (579-1124). Thirteen of h19 patients (11/15 boys and 2/4 girls) had an ICV between ±2 SD curves of Lichtenberg, 2 of 19 (1/15 boys and 1/4 girls) had an ICV less than −2 SD and 4 of 19 (3/15 boys and 1/4 girls) had an ICV greater than +2 SD. Mean UCSQ severity of trigonocephaly was 2.40 (−622.65 to 1279.75). Correlation between severity and ICV was negligible (r = −0.11). No papilledema was reported.Conclusions:Measured ICV was within normal ranges for trigonocephaly patients, in both mild and severe cases. No correlation was found between severity of trigonocephaly and ICV.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-17T09:04:32Z
      DOI: 10.1177/10556656211025185
       
  • Awareness of Orofacial Clefts in the Sultanate of Oman

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      Authors: Khalifa Al Alawi, Sultan Al Shaqsi, Mouzan Al Ghaithi
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:Cleft lip and/or palate (CL/P) is the most common craniofacial anomaly. In Oman, the incidence of CL/P is estimated to be 1.5 per 1000 live births per year. Knowledge, awareness, and attitude toward CL/P help to optimize the management of the condition and reduce social and emotional stress among this group of children and their family. In this survey, we assessed the public awareness, knowledge, and attitude toward this deformity in the Sultanate of Oman.Method:A cross-sectional study was conducted using validated questionnaire that was distributed using Google forms.Result:A total of 739 participants completed the survey. The majority is aware of cleft lip (86.7%) and palate (63.2%). In general, attitude toward patients with CL/P was positive. Females had higher knowledge compared to males, 54.4% and 32.9%, respectively. Online resources were the main source of information.Conclusion:There are variations in knowledge, awareness, and attitude toward CL/P among participants. Omani population needs to be educated more about CL/P, its complications if not treated, and the available treatment.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-17T09:03:31Z
      DOI: 10.1177/10556656211024477
       
  • Utilizing Teledentistry to Manage Cleft Lip and Palate Patients in an
           Outpatient Setting

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      Authors: Asvitha Viswanathan, Nikita Patel, Mina Vaidyanathan, Nabina Bhujel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:As a result of the current global pandemic, the dental profession has utilized teledentistry to reduce footfall in the hospitals and clinics where possible. Pediatric dental consultants form a vital part of a multidisciplinary team and regularly monitor the dental growth and development of patients with cleft lip and palate.Objective:To assess the effectiveness of the service provided by pediatric dental consultants in the South Thames Cleft Service at Evelina Children’s Hospital during the COVID-19 pandemic through virtual clinics.Design:Data were collected retrospectively and include all cleft patients contacted via the virtual clinic during May to July 2020. Patients were prioritized by the Red, Amber, Green (RAG) scale to highlight the urgency of their next face-to-face appointment.Results:A total of 215 patients were contacted during this period with a 97% response rate. Patients given a RAG score of GREEN (86%) meant no urgent requirement for a face-to-face consultation and AMBER (8%) patients required treatment that was deemed nonurgent. However, 3% of patients received a RED rating as they required urgent input.Conclusion:Through these virtual clinics, the pediatric team was able to reach 208 patients and provided advice and reassurance. The need for face-to-face appointment was eliminated for 11% of patients who were discharged to their local dental practitioners, thereby reducing the risk of spreading COVID-19.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-17T09:02:52Z
      DOI: 10.1177/10556656211023244
       
  • Through-and-Through Dissection of the Soft Palate for Pharyngeal Flap
           Inset: A “Good-Fast-Cheap” Technique for Any Etiology of
           Velopharyngeal Incompetence

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      Authors: Michael Carr, Michaela Skarlicki, Sheryl Palm, Marija Bucevska, Jeffrey Bone, Arun K. Gosain, Jugpal S. Arneja
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To determine the efficacy and resource utilization of through-and-through dissection of the soft palate for pharyngeal flap inset for velopharyngeal incompetence (VPI) of any indication.Design:Retrospective review.Setting:Tertiary care center.Patients:Thirty patients were included. Inclusion criteria were diagnosis of severe VPI based on perceptual speech assessment, confirmed by nasoendoscopy or videofluoroscopy; VPI managed surgically with modified pharyngeal flap with through-and-through dissection of the soft palate; and minimum 6 months follow-up. Patients with 22q11.2 deletion syndrome were excluded.Intervention:Modified pharyngeal flap with through-and-through dissection of the soft palate.Main Outcome Measure(s):Velopharyngeal competence and speech assessed using the Speech-Language Pathologist 3 scale.Results:The median preoperative speech score was 11 of 13 (range, 7 to 13), which improved significantly to a median postoperative score of 1 of 13 (range 0-7; P < .001). Velopharyngeal competence was restored in 25 (83%) patients, borderline competence in 3 (10%), and VPI persisted in 2 (7%) patients. Complications included 1 palatal fistula that required elective revision and 1 mild obstructive sleep apnea that did not require flap takedown. Median skin-to-skin operative time was 73.5 minutes, and median length of stay (LOS) was 50.3 hours.Conclusions:This technique allows direct visualization of flap placement and largely restores velopharyngeal competence irrespective of VPI etiology, with low complication rates. Short operative time and LOS extend the value proposition, making this technique not only efficacious but also a resource-efficient option for surgical management of severe VPI.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-17T09:01:43Z
      DOI: 10.1177/10556656211021738
       
  • Determination of Ethnic Variation in Infant Nasolabial Anthropometry Using
           3D Photographs: Implications for Bilateral Cleft Lip Nasal Correction

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      Authors: Diana S. Jodeh, Jacqueline M. Ross, Maria Leszczynska, Fatima Qamar, Rachel L. Dawkins, James J. Cray, S. Alex Rottgers
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:We aimed to assess significant ethnic variabilities in infants’ nasolabial anthropometry to motivate variations in surgical correction of a synchronous bilateral cleft lip/nasal anomaly, specifically whether a long columella is a European feature, therefore accepting a short columella and/or delayed columellar lengthening suitable for reconstruction in ethnic patients.Methods:Thirty-three infants without craniofacial pathology (10 African American [AA], 7 Hispanic [H], and 16 of European descent [C]), ages 3 to 8 months, presenting to the Johns Hopkins All Children’s general pediatric clinic were recruited. Four separate 3D photographs (2 submental and frontal views each) were taken using the Vectra H1 handheld camera (Canfield Imaging). Eighteen linear facial distances were measured using Mirror 3D analysis (Canfield Imaging Systems). Difference between ethnicities was measured using analysis of variance with the Bonferroni/Dunn post hoc comparisons. Pearson correlation was employed for interrater reliability. All statistical analyses were carried out using SPSS version 21.0 (IBM Corp), with statistical significance set at P < .05.Results:Nasal projection (sn-prn) and columella length (sn-c) did not differ significantly between groups (P = .9). Significant differences were seen between ethnic groups in nasal width (sbal-sbal [C-AA; P = .02]; ac-ac [C-AA; P = .00; H-AA; P = .04]; al-al [C-AA; P = .00; H-AA; P = .001]) and labial length (sn-ls [C-AA; P = .041]; sn-sto [C-AA; P = .005]; Cphs-Cphi L [C-AA; P = .013]; Cphs-Cphi R [C-AA; P = .015]). Interrater reliability was good to excellent and significantly correlated for all measures.Conclusions:African American infants exhibited wider noses and longer lips. No difference was noted in nasal projection or columella length, indicating that these structures should be corrected during the primary cleft lip and nasal repair for all patients and should not be deferred to secondary correction.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-16T09:54:11Z
      DOI: 10.1177/10556656211024470
       
  • Photometric Evaluation of Adult Patients With Bilateral Cleft Lip and
           Palate Treated With Nasoalveolar Molding and Primary Columella Lengthening
           

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      Authors: Maria Costanza Meazzini, Francesca Parravicini, Vera Donati, Roberto Brusati, Federico Biglioli, Luca Autelitano
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      A short columella and a flattened nasal tip are the characteristic stigmata of patients with complete bilateral cleft lip and palate (BCLP).Objective:The aim of this study was to assess the nasal shape of young adults with BCLP treated with primary surgical columella lengthening and nasoalveolar molding (NAM).Setting and Patients:A group of 28 young adult patients with BCLP (mean age: 19.1±1.4 years) was compared through normalized photogrammetry to a control of 28 age- and sex-matched noncleft young adults.Results:Nasal protrusion and length of the columella were not different from noncleft young adults. On the other hand, nasolabial angle, columellar width, interalar, and nasal tip width were significantly wider than the noncleft controls. Thus, 27% of the patients have requested at this time secondary correction of the excessive nasal width.Conclusions:Both NAM and primary rhinoplasty in patients with BCLP resulted in a near normal length of the columella and nasal projection until young adulthood. Nevertheless, width of all nasal features was significantly wider than the noncleft population and required secondary nasal correction in one-third of the sample.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-16T09:53:15Z
      DOI: 10.1177/10556656211024069
       
  • Evaluation of Facial Appearance–Related Quality of Life in Young
           Japanese Patients With Cleft Lip and/or Palate

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      Authors: Ayaka Oka, Chihiro Tanikawa, Yukako Isogai, Kiyomi Mihara, Takashi Yamashiro
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:(1) To confirm the reliability of a Japanese version of the Youth Quality of Life Instrument-Facial Differences Module (YQOL-FD); (2) to assess the quality of life (QoL) related to facial difference in Japanese youths with cleft lip and/or palate (CL/P) using this instrument; and (3) to examine the QoL differences according to age, sex, and cleft type.Design:A cross-sectional study.Setting:Japanese youths with CL/P were recruited through our hospital and asked to complete the YQOL-FD.Participants:Sixty-nine Japanese youths (age, 11-18 years) with CL/P.Outcome Measures:The domain scores of stigma, negative consequences, negative self-image, positive consequences, and coping in the YQOL-FD, and the reliability of such scores were evaluated.Results:The instrument showed an acceptable internal consistency (Cronbach α = 0.74-0.92) and test–retest reliability (intraclass correlation coefficient = 0.94-0.98), except for the coping domain. The individual’s domain scores were spread out from the lowest score to the high scores among all domains, thus indicating the negative and positive impacts of living with facial differences regarding their QoL may vary among individuals with CL/P. All domain scores in the 15- to 18-year-old group were significantly higher than those in 11- to 14-year-old group; there were no significant differences according to sex or cleft type.Conclusions:The instrument showed acceptable reliability, except for the coping domain. There were individual variations in QoL concerning the facial difference among Japanese youths with CL/P as measured by the YQOL-FD, suggesting the importance of individual evaluations. Perceptions were influenced by age, but not sex or cleft type.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-16T09:47:09Z
      DOI: 10.1177/10556656211023243
       
  • Morphometric Variations and Growth Analysis of the Side Face Profiles of
           Chinese Children

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      Authors: Haijun Li, Huimin Chen, Liming Liu, Yujie Zhao
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:We aim to use geometric morphometric methods to analyze the contours of the side face profile and its variations in 116 children in Hunan province, China, in order to reveal common morphological features and growth of the side face profiles of Chinese children.Design:We photographed the side faces with consistent standardized procedures. Thirty-seven landmarks were recorded, which comprehensively reflected the facial sagittal plane. The collection of coordinate data regarding landmarks was conducted by tpsDig software. Other analyses, such as average shape analysis, principal component analysis, variations analysis, and allometric analysis, were conducted using tpsRelw and tpsRegr.Result:Based on the 37 landmarks, principal component analysis was used on the profiles of boys and girls. The result shows that PC1 and PC2 account for 35.46% and 27.44% of the side face contour variations, respectively. There is no significant difference in the side profiles of boys and girls. Overlapping distributions between the boys and girls occurred. A significant difference occurs when principal component analysis was used on the side profiles of children and adults.Conclusions:There is a significant difference in the side face profiles between adult males and females. Adult males have a higher brow (the 8th landmark) than females, and adult females have a more curved forehead. However, the side face profiles of boys and girls around 10 years of age are generally similar, which suggests that the significant difference in side face profiles between adult males and females may be formed after the age of 10. This study is of great significance for both orthodontists and pediatricians to use different norms for kids and adults and different norms for males and females for each of those age groups.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-15T09:18:15Z
      DOI: 10.1177/10556656211022919
       
  • Feasibility of Social Media Recruitment for Orofacial Cleft Genetic
           Research

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      Authors: Grace Carlock, Kelly Manning, Elizabeth J. Leslie
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:This study assessed the feasibility of unpaid social media advertising to recruit participants affected with an orofacial cleft (OFC) for a genetic study.Design:This is a retrospective analysis of recruitment based on enrollment and participation in a genetic study. Participants completed a series of enrollment surveys, provided saliva samples, and completed postparticipation feedback surveys.Participants:Participants were eligible if they or a minor in their care were affected by an OFC, the affected participant was not adopted, and the mother of the affected individual had not taken antiseizure medication during pregnancy.Main Outcome Measures:Success of recruitment was evaluated from the number of enrolled participants and sample return rate.Results:In the first 12 months of recruitment, 313 individuals completed initial screening surveys; of these, 306 participants were eligible. A total of 263 individuals completed all online surveys and were sent DNA sample kits. One hundred sixty-two subject DNA samples were returned within 12 months of sending, for a return rate of 62%. Approximately two-thirds (66.3%) of all returned samples were sent back within the first 6 weeks after receiving DNA kits.Conclusions:Unpaid social media advertising enabled the recruitment of a large cohort of participants in a short time (12 months). The resulting study population was limited in racial and ethnic diversity, suggesting that other recruitment strategies will be needed for studies seeking specific demographic or socioeconomic groups. Nonetheless, social media recruitment was efficient and effective for recruiting participants for a genetic study in comparison to traditional clinic-based modes of recruitment.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-14T09:11:54Z
      DOI: 10.1177/10556656211024484
       
  • Pre-Hispanic Evidence of Cleft Lip and/or Palate in Mesoamerica

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      Authors: Ramon Manuel Aleman, Maria Guadalupe Martinez
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      In this letter, the authors present archeological evidence of the presence of cleft lip and/or palate (CLP) in pre-Hispanic Mesoamerica. During years 2016 and 2017, the authors visited 5 anthropology museums in Mexico, Guatemala, and El Salvador, in search for pre-Hispanic archeological evidence of CLP, and 16 anthropomorphic figurines with evidence of CLP were identified; 9 at the Anahuacalli Museum, 6 at the National Anthropology Museum of Mexico, and 1 at the National Anthropology Museum of El Salvador. Fifteen of these ceramics originated from the shaft tombs (pre-Hispanic culture from western Mexico, Jalisco, Colima, and Nayarit; dating from 200 bc-600 ad), and 1 ceramic originated from the Cotzumalguapa (a pre-Hispanic culture from western El Salvador dating from 200 ad-900 ad).
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-11T09:31:34Z
      DOI: 10.1177/10556656211021701
       
  • A Closer Look at Delayed Primary Cleft Surgery and Unrepaired Cleft Lip
           and/or Palate in 5 UK Cleft Centers

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      Authors: Sophie Butterworth, Clare Rivers, Marnie Fullarton, Colm Murphy, Victoria Beale, Jason Neil-Dwyer, Simon Van Eeden, Stephanie Van Eeden, Peter D. Hodgkinson, Alistair Smyth, David C Sainsbury
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:There may be many reasons for delays to primary cleft surgery. Our aim was to investigate the age of children undergoing primary cleft lip or primary cleft palate repair in 5 cleft centers within the United Kingdom. Identify the reasons for delayed primary cleft lip repair (beyond 6 months) and delayed primary palate repair (beyond 13 months). Identify children who had a cleft lip and/or palate (CL±P) that was intentionally unrepaired and the reasons for this.Methods:A retrospective, multicenter review of patients born with a CL±P between December 1, 2012, and December 31, 2016. Three regional cleft centers, comprising of 5 cleft administrative units in the United Kingdom participated.Results:In all, 1826 patients with CL±P were identified. Of them, 120 patients had delayed lip repair, outside the expected standard of 183 days. And, 178 patients in total had delayed palate repair, outside the expected standard of 396 days. Twenty (1%) patients had an unrepaired cleft palate.Conclusions:This large retrospective review highlights variations between centers regarding the timing of lip and palate surgery and details the reasons stated for delayed primary surgery. A small number of patients with an unrepaired cleft palate were identified. All had complex medical problems or comorbidities listed as a reason for the decision not to operate and 50% had a syndromic diagnosis. The number of patients receiving delayed surgery due to comorbidities, being underweight or prematurity, highlights the importance of the cleft specialist nurse and pediatrician within the cleft multidisciplinary team.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-10T09:31:52Z
      DOI: 10.1177/10556656211021700
       
  • ALX-Related Frontonasal Dysplasias: Clinical Characteristics and Surgical
           Management

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      Authors: Ibrahim Vargel, Halil Ibrahim Canter, Arda Kucukguven, Asim Aydin, Figen Ozgur
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Aim:The term frontonasal dysplasia (FND) represents a spectrum of anomalies and its genetics have not been well defined. Recently, the critical role of the aristaless-like homeobox (ALX) gene family on the craniofacial development has been discovered. In the present study, we aimed to propose a systematic surgical treatment plan for the ALX-related FNDs according to the genotypic classification as well as demonstrating their clinical characteristics to help surgeons diagnose the underlying pathology accurately.Design:Single-institution retrospective.Setting:Tertiary health care.Patients and Methods:Eighty-nine FND cases were evaluated. Eight of them had ALX1-related FND3, 3 had ALX3-related FND1, and 2 had ALX4-related FND2. Phenotype characteristics of ALX-related FNDs were evaluated, and relevant surgical interventions were assessed.Results:The ALX1-related FND3 phenotype is striking due to the involvement of the eyes in addition to the presence of hypertelorism, facial clefts, and nasal deformities. A widened philtrum and prominent philtral columns are remarkable features of the ALX3-related FND1, whereas the ALX4-related FND2 has more severe deformities: severe hypertelorism, brachycephaly, large parietal bone defects, broad nasal dorsum, and alopecia. Facial bipartition, box osteotomies, eyelid coloboma repair, cleft lip and palate repair, nasal reconstruction, and fronto-orbital advancement can be performed in ALX-related FNDs based on the characteristics of each subtype.Conclusions:This genetic classification system will help surgeon diagnose patients with FND with unique features and draw a roadmap for their treatment with a better surgical perspective.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-08T08:50:39Z
      DOI: 10.1177/10556656211019621
       
  • Mechanical Tension-Stress in Alveolar Cleft Repaired With Autogenous Bone
           in Canine Models

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      Authors: Anqi Liu, Jialiang Huang
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Cleft lip and/or palate is a common birth defect worldwide, always accompanied by alveolar cleft. However, the success rate of secondary alveolar bone grafting is unsatisfactory. Rapid maxillary expansion (RME) often used after bone transplantation provides functional stimulation for bone graft area. This study aimed to investigate the effect of RME force on the bone graft area and midpalatal suture, and screen out the most suitable loaded force and loaded teeth, so as to provide a reference for clinical treatment.Methods:Fourteen 24-week-old male beagles were assigned randomly to 3 groups: blank control, autogenous, and autogenous with RME. Three-dimensional finite element analysis was conducted to evaluate the distribution and value of the stress in the model. The maxillae were collected and subjected to radiography and helical computed tomography to evaluate new bone formation in the graft area. Van Gieson’s Picrofuchsin staining was performed for histomorphological observation.Results:After 8 weeks of RME treatment, new bone formation of the dogs was markedly accelerated, and bone resorption was significantly reduced compared with the untreated dogs or those only treated with autogenous iliac bone. The treatment with RME evidently made the bone trabecula more abundant and the area of bone formation larger. Three-dimensional finite element analysis showed that the clinical effect can be achieved by using canine teeth as the loaded teeth and applying force of 10 MPa.Conclusion:Rapid maxillary expansion after bone grafting had a positive effect on osteogenesis in a canine model of alveolar cleft.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-08T08:41:26Z
      DOI: 10.1177/10556656211018950
       
  • Exploring the Relationship Between Palatal Cleft Type and Width With the
           Use of Relieving Incisions in Primary Repair

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      Authors: Alex Davies, Amy Davies, Yvonne Wren, Scott Deacon, Alistair R.M. Cobb, Shaheel Chummun
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:The mainstay of palatal repair in the United Kingdom is the intravelar veloplasty (IVVP). It is not always possible to align the oral mucosa in the midline to achieve tension-free repair. The addition of lateral relieving incisions may aid transposition of the oral mucosa to allow closure. The aim of this study was to explore cleft features that may predispose to a requirement for relieving incisions in order to allow palate closure.Design:We performed a national multiinstitutional retrospective study using data from the UK Cleft Collective cohort study.Patients:The study sample consisted of 474 patients who had undergone IVVP at the time of palatal closure across all 16 of the UK cleft units.Results:We found strong evidence for the requirement for relieving incisions in patients with an increased degree of clefting per the Veau classification (P < .001), increasing palatal soft-edge width (P < .001) and moderate evidence of an associated use in patients with Pierre Robin sequence (P = .015). Insufficient data were available to explore the relationship between intertuberosity distance and the presence of fistula formation with the use of relieving incisions.Conclusions:The results of this study identify cleft features that increase the likelihood for requiring lateral relieving incisions to allow palatal closure. The degree to which the addition of relieving incisions to IVVP affects maxillary growth and speech outcomes is unknown. Further study is required to answer this important question.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-04T09:50:57Z
      DOI: 10.1177/10556656211019616
       
  • Speech Outcomes Following Orticochea Pharyngoplasty in Patients With
           History of Cleft Palate and Noncleft Velopharyngeal Dysfunction

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      Authors: Alison L. Birch, Zoe V. Jordan, Louisa M. Ferguson, Clare B. Kelly, John G. Boorman
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To report speech outcomes following Orticochea pharyngoplasty in 43 patients with cleft palate and noncleft velopharyngeal dysfunction.Design:A retrospective surgical audit of patients undergoing Orticochea pharyngoplasty between 2004 and 2012, with speech as a primary outcome measure.Setting:Patients known to a regional UK cleft center.Methods:Forty-three patients underwent Orticochea pharyngoplasty by a single surgeon in a UK regional cleft center. Twenty-one patients had undergone a prior procedure for velopharyngeal dysfunction. Pre- and postoperative speech samples were assessed blindly using the Cleft Audit Protocol for Speech-Augmented by a specialist cleft speech and language therapist, external to the team. Speech samples were rated on the following parameters: hypernasality, hyponasality, audible nasal emission, nasal, turbulence, and passive cleft speech characteristics. Statistical differences in pre- and postoperative speech scores were tested using the Wilcoxon matched-pairs signed-ranks test. Inter- and intrareliability scores were calculated using weighted Cohen κ.Results:Whole group: A statistically significant difference in pre- and postoperative scores for hypernasality (P < .001), hyponasality (P < .05), nasal emission (P < .01), and passive cleft speech characteristics (P < .01) were reported. Patients with cleft diagnoses: A statistically significant difference in scores for hypernasality (P < .001), nasal emission (P < .01), and passive cleft speech characteristics (P < .01) were reported for this group of patients. Patients with noncleft diagnoses: The only parameter to demonstrate a statistically significant difference was hypernasality (P < .01) in this group.Conclusions:Orticochea pharyngoplasty is a successful surgical procedure in treating velopharyngeal dysfunction in both the cleft and noncleft populations.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-04T09:48:07Z
      DOI: 10.1177/10556656211010623
       
  • A Systematic Review of Mandibular Distraction Osteogenesis Versus
           Orthodontic Airway Plate for Airway Obstruction Treatment in Pierre Robin
           Sequence

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      Authors: Darren B. Abbas, Christopher Lavin, Evan J. Fahy, HyeRan Choo, Mai Thy Truong, Karl C. Bruckman, Rohit K. Khosla, H. Peter Lorenz, Arash Momeni, Derrick C. Wan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Mandibular distraction osteogenesis (MDO) is frequently performed to address airway obstruction in patients with Pierre Robin sequence (PRS), though more recently the technique of orthodontic airway plating (OAP) has gained traction. We aimed to evaluate OAP compared to MDO for airway obstruction in PRS.Design:A systematic literature search across PubMed, Embase, and Google Scholar identified all studies published in English, which involved MDO or any form of OAP as treatments for PRS. All relevant articles were reviewed in detail and reported on, adhering to PRISMA guidelines.Main Outcome Measures:Airway (tracheostomy avoidance, decannulation rate), feeding (full oral feeding tolerance).Results:Literature search identified 970 articles, of which 42 MDO studies and 9 OAP studies met criteria for review. A total of 1159 individuals were treated with MDO, and 322 individuals were treated with OAP. Primary outcomes appear similar for MDO and OAP at face value; however, this must be interpreted with different pretreatment contexts in mind.Conclusions:Orthodontic airway plating may be considered for airway obstruction in PRS, as some airway-related and feeding-related outcomes appear similar with MDO, per existing evidence in the literature. However, since PRS severity differed between studies, OAP cannot be uniformly considered a replacement for MDO. Further research is required to more comprehensively assess these treatment modalities inclusive of metrics that allow for direct comparison.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-06-02T09:03:41Z
      DOI: 10.1177/10556656211011886
       
  • The International Family Study of Nonsyndromic Orofacial Clefts: Design
           and Methods

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      Authors: Allyn Auslander, Roberta McKean-Cowdin, Devin Feigelson, Frederick Brindopke, Melissa DiBona, Kathy Magee, Lili Arakaki, Rijuta Kapoor, Stephanie Ly, David V. Conti, Sylvia Rakotoarison, Fouzia Mahmoudi, Albertina McGregor, Melissa Giron, Angela Rose Hernandez, Thi-Hai-Duc Nguyen, Anselme Mwepu, Pedro A. Sanchez-Lara, William Magee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:The majority of research to understand the risk factors of nonsyndromic orofacial clefts (NSOFCs) has been conducted in high-income populations. Although patients with NSOFCs in low- and middle-income countries (LMICs) are at the highest risk of not receiving care, global health infrastructure allows innovative partnerships to explore the etiologic mechanisms of cleft and targets for prevention unique to these populations.Methods:The International Family Study (IFS) is an ongoing case–control study with supplemental parental trio data designed to examine genetic, environmental, lifestyle, and sociodemographic risk factors for NSOFCs in 8 LMICs (through August 2020). Interview and biological samples are collected for each family. The interview includes demographics, family history of cleft, diet and water sources, maternal pregnancy history, and other lifestyle and environmental factors.Results:Seven of 8 countries are currently summarized (2012-2017) for a total of 2955 case and 2774 control families with 11 946 unique biological samples from Vietnam, Philippines, Honduras, Madagascar, Morocco, Democratic Republic of the Congo, and Nicaragua. The phenotype distribution was 1641 (55.5%) cases with cleft lip and palate, 782 (26.5%) with cleft lip (CL), and 432 (14.6%) with cleft palate (CP).Discussion:The International Family Study is the largest case set of NSOFCs with an associated biobank in LMICs currently assembled. The biobank, family, and case–control study now include samples from 8 LMICs where local health care infrastructure cannot address the surgical burden of cleft or investigate causal mechanisms. The International Family Study can be a source of information and may collaborate with local public health institutions regarding education and interventions to potentially prevent NSOFCs.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-31T09:25:40Z
      DOI: 10.1177/10556656211018956
       
  • Cleft Lip and/or Palate and Associated Risks in Lower-Middle-Income
           Countries: A Systematic Review

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      Authors: Kayla Kruppa, Esedra Krüger, Carlien Vorster, Jeannie van der Linde
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To identify and review published data on the risks associated with cleft lip and/or palate (CL/P) in lower-middle-income countries (LMICs).Design:A systematic review of literature was performed on electronic databases using the Preferred Reporting Items for Systematic Reviews and Meta-Analysis Protocols. Literature on risks associated with CL/P in LMICs, from 2010 to 2020, were included.Results:Seventeen studies met the inclusion criteria. All studies adopted an observational study design. Biological and environmental risks were identified. Maternal and paternal age (n = 7) and low socioeconomic status (n = 5) were the most prominently associated environmental risk factors. A strong association was identified between family history of cleft (n = 7) and CL/P occurrence.Conclusion:Environmental risk factors are now being investigated more than biological risk factors in LMICs, aiding health care workers in the early identification of possible cumulative effects of risks in CL/P. Contextually relevant tools are recommended to promote early identification of at-risk infants.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-31T09:25:10Z
      DOI: 10.1177/10556656211018952
       
  • Three-Dimensional Nasolabial Changes After Nasoalveolar Molding and
           Primary Lip/Nose Surgery in Infants With Bilateral Cleft Lip and Palate

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      Authors: Laura Mancini, Shayna Avinoam, Barry H. Grayson, Roberto L. Flores, David A. Staffenberg, Pradip R. Shetye
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Utilize 3-dimensional (3D) photography to evaluate the nasolabial changes in infants with bilateral cleft lip and palate (BCLP) who underwent nasoalveolar molding (NAM) and primary reconstructive surgery.Design:This is a retrospective serial longitudinal study of consecutively enrolled infants from September 2012 to July 2016 with BCLP who underwent NAM before primary lip and nose reconstructive surgery. It included infants who had digital 3dMD stereophotogrammetry records at initial presentation (T1), completion of NAM (T2), and 3 weeks following primary repair (T3). Twelve infants fulfilled the inclusion criteria. 3dMD Vultus software was used to orient images and plot 16 nasolabial points with x, y, z coordinates to obtain the linear and angular measurements. Nasal form changes were measured and analyzed between T1 (0.5 months old), T2 (5 months old), and T3 (6 months old). Intraclass correlation coefficient was performed for intrarater reliability. Averaged data from the 3D images was statistically analyzed from T1 to T2 and T2 to T3 with Wilcoxon tests. Unaffected infant norms from the Farkas publication were used as a control sample.Results:After NAM therapy, statistically significant changes in the position of subnasale and labius superius improved nasolabial symmetry. Both retruded after NAM were displaced downward after NAM and surgical correction with respect to soft tissue nasion. The nasal tip’s projection was maintained with NAM and surgical correction. The columella lengthened from 1.4 to 4.71 mm following NAM.Conclusions:There was a significant improvement in the nasolabial anatomy after NAM, and this was further enhanced after primary reconstructive surgery.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-25T09:15:52Z
      DOI: 10.1177/10556656211012858
       
  • Treatment of Cleft Lip and Palate in the Amazon Region in Brazil: Speech
           Results and Sociodemographic Aspects

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      Authors: Laryssa Lopes de Araújo, Nivaldo Alonso, Ana Paula Fukushiro
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To investigate the speech outcomes in individuals with cleft lip and palate (CLP) from the Amazon region and determine their correlation with sociodemographic factors.Design:Cross-sectional, prospective, single-blind study.Setting:Tertiary care institute in Manaus, Amazonas, Brazil.Patients:Four hundred twenty individuals with CLP, aged 4 to 57 years.Main Outcome Measures:The samples were judged by 3 speech pathologists experienced in the speech assessment of individuals with CLP. Hypernasality was scored using a 4-point scale, being 1 = absent, 2 = mild, 3 = moderate, and 4 = severe, and the active speech symptoms were classified as absent or present. The final score for each speech symptom was reached by consensus among the 3 examiners Descriptive analysis of sociodemographic data included origin, socioeconomic status, type of cleft, surgical technique employed, the age they underwent primary and secondary palatoplasty, and palatal fistula.Results:Absence of hypernasality was observed in 41% of the individuals, 18% had mild, 28% moderate, and 13% severe hypernasality. Active speech symptoms were observed in 57% of the individuals. Significant correlations were found between speech outcomes and the variables such as origin, socioeconomic status, age at primary and secondary palatoplasty, and presence of fistula.Conclusions:Most individuals with repaired CLP from the Amazon region presented speech disorders, characterized by hypernasality and active errors. Patients living outside the state capital, of low socioeconomic level, underwent palatoplasty late and with presence of palatal fistula tended to have the worst speech outcomes.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-25T08:55:59Z
      DOI: 10.1177/10556656211017791
       
  • How Does Hypodontia Compare in Nonsyndromic Pierre Robin Sequence Versus
           Isolated Cleft Palate and Isolated Cleft Lip'

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      Authors: Maria Dillon, Madhavi Seshu, Norah Flannigan, Susana Dominguez-Gonzalez
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To assess the prevalence and patterns of hypodontia in nonsyndromic Pierre Robin sequence (PRS) and compare it with hypodontia in nonsyndromic isolated cleft palates and isolated cleft lips.Design:Retrospective cohort study.Setting:Alder Hey Children’s Hospital, United Kingdom.Patients:Patients with nonsyndromic PRS (group 1), isolated cleft palate (group 2), and isolated cleft lip (group 3).Main Outcome Measures:Hypodontia in the permanent dentition assessed from orthopantomographs.Results:A total of 154 patients were included. Group 1 had the highest incidence of hypodontia with 47% having at least one tooth congenitally absent. Groups 2 and 3 had reduced rates of hypodontia with 27% and 19% of the groups missing teeth, respectively; 93% of cases of hypodontia in group 1 involved the absence of at least one second premolar. Of these patients, there was found to be bilateral agenesis of second premolars in 50% of cases.Conclusions:Patients with PRS and cleft palates are more likely to have hypodontia than those with isolated cleft palates or unilateral cleft lips. Patients with PRS have more severe hypodontia than those with isolated cleft palates or unilateral cleft lips. Bilateral agenesis of lower second premolars is a commonly seen pattern among patients with PRS. In this large UK study, a similar prevalence and pattern of hypodontia to other nonsyndromic PRS populations worldwide has been demonstrated.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-21T09:33:34Z
      DOI: 10.1177/10556656211017778
       
  • Repair of Asymmetric Bilateral Cleft lip: 1 Stage or 2 Stage

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      Authors: Xinran Zhao, Yilai Wu, Guomin Wang, Yusheng Yang, Ming Cai
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To verify the advantages and indications of 1-stage and 2-stage repair for asymmetric bilateral cleft lip (BCL).Design:Retrospective study.Setting:From January 2004 to December 2016 in our department.Patients:Patients with BCL.Main Outcome Measure(s):Over 6 months after the operation, the surgery outcomes were evaluated and graded by 2 experienced surgeons.Results:The result of surgery was evaluated using the scoring method of Mortier et al and Anastassov and Chipkov. Among 133 patients with asymmetric BCL, 61 (45.9%) had 1-stage repair and 72 (54.1%) had 2-stage repair. Sixty-eight (51.1%) patients had complete-incomplete cleft lip (CL), and those who underwent 1-stage repair showed a trend of better outcome (P = .028). Fifty (37.6%) patients with incomplete-microform CL showed no significant difference between the outcomes of 2 surgery plans (P = .253). In 15 (11.3%) patients with complete-microform CL, only one had 1-stage repair with a score of 8.5. The other 14 patients with 2-stage repair were scored 3.68 ± 1.28. Two-stage repair was preferable when the deformity degree was very different on 2 sides, as it could reduce unnecessary scar tissue and extend the nasal columella. One-stage repair could help to achieve the anatomical reduction of the orbicularis oris and a better contour of the vermilion tubercle.Conclusion:One-stage repair is recommended for patients with complete-incomplete CL and incomplete-microform CL. Two-stage repair for patients with complete-microform CL is preferred in our center, but more studies are required to support this conclusion.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-21T09:25:33Z
      DOI: 10.1177/10556656211013978
       
  • Knowledge and Attitude Among Various Dental Specialities Regarding
           Nasoalveolar Molding Procedure in Cleft Patients—A Cross-Sectional Study
           

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      Authors: Divya Doddamani, Saraswathi V. Naik, Archana P. Betur, Sugandhan Suriyan, Basappa Nadig
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:Nasoalveolar molding (NAM) is a presurgical orthopedic procedure, mainly focusing upon the aesthetic outcome of the treatment goal by reducing the number of reconstructive surgeries performed later for the purpose of esthetics. It was reported a very small proportion of pediatric dentists are providing presurgical intervention and the practice of NAM still side stepping among various dental practitioners.Objectives:The study was aimed to assess the knowledge and attitude among various dental specialties regarding NAM procedure in cleft patients.Materials and Methods:A questionnaire-based cross-sectional survey was carried out among 180 dental professionals (pedodontists, orthodontists, prosthodontists, oral surgeons). A total of 180 questionnaires were prepared, out of which 98 were filled through direct personal interview and 82 were circulated via Google forms. Results were analyzed using chi-square test, Mann-Whitney U test, and Kruskal-Wallis test.Results:There was statistically significant difference in the cumulative knowledge scores between specialty with P < .001; 99% of the participants think one should opt for NAM before cleft lip and palate surgery and 39% participants prefer pedodontists to carry out NAM procedure.Conclusion:Dental specialists in India have a positive attitude toward presurgical orthopedics. Majority of them agreed that there is great improvement in the aesthetic outcomes of surgeries after NAM intervention.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-20T09:07:07Z
      DOI: 10.1177/10556656211017404
       
  • Otitis Media and Hearing Loss in Patients With Nonsyndromic
           Craniosynostosis: A Multicenter Study

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      Authors: Jeewanjot S. Grewal, Jason E. Cohn, Jacob Burdett, Alex Tampio, Jordan Licata, Wellington J. Davis, Sherard A. Tatum, Brian D. Nicholas
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:The objectives of this study were to: (1) determine the prevalence of otitis media with effusion in patients with nonsyndromic craniosynostosis; (2) determine the prevalence of hearing loss in patients with nonsyndromic craniosynostosis; and (3) identify potential patterns and outcomes in patients with nonsyndromic craniosynostosis.Methods:A retrospective chart review was conducted at 2 academic institutions, St Christopher’s Hospital for Children and SUNY Upstate Medical University, from January 2015 through August 2018, to identify patients having nonsyndromic craniosynostosis with a concurrent diagnosis of otitis media and/or hearing loss. The demographic data and categorical variables were analyzed using descriptive statistics and chi-square testing, respectively.Results:In the entire cohort of patients (N = 113, age range 0-123 months), 36% had otitis media with effusion on either history, physical examination, tympanometry, and/or imaging. Half (50%) of patients with coronal synostosis had otitis media with effusion compared to sagittal (40.7%), metopic (26.3%), multiple (25%), and lambdoid (0%). However, these differences were not statistically significant (P = .190). Most patients had normal hearing (91%), while a minority had either conductive (7%) or sensorineural (2%) hearing loss.Conclusion:The presence of otitis media in our cohort of patients with nonsyndromic craniosynostosis appears to be at the upper limit of normal when compared to historical rates in normocephalic children. Synostosis subtype did not appear to predict the presence of otitis media. Only 9% of patients with nonsyndromic craniosynostosis were found to have a hearing loss.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-18T09:00:37Z
      DOI: 10.1177/10556656211017795
       
  • Optimizing Outcomes After Cleft Palate Repair: Design and Implementation
           of a Perioperative Clinical Care Pathway

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      Authors: Thomas R. Cawthorn, Anna R. Todd, Nina Hardcastle, Adam O. Spencer, A. Robertson Harrop, Frankie O. G. Fraulin
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate the development process and clinical impact of implementing a standardized perioperative clinical care pathway for cleft palate repair.Design:Medical records of patients undergoing primary cleft palate repair prior to pathway implementation were retrospectively reviewed as a historical control group (N = 40). The historical cohort was compared to a prospectively collected group of patients who were treated according to the pathway (N = 40).Patients:Healthy, nonsyndromic infants undergoing primary cleft palate repair at a tertiary care pediatric hospital.Interventions:A novel, standardized pathway was created through an iterative process, combining literature review with expert opinion and discussions with institutional stakeholders. The pathway integrated multimodal analgesia throughout the perioperative course and included intraoperative bilateral maxillary nerve blocks. Perioperative protocols for preoperative fasting, case timing, antiemetics, intravenous fluid management, and postoperative diet advancement were standardized.Main Outcome Measures:Primary outcomes include: (1) length of hospital stay, (2) cumulative opioid consumption, (3) oral intake postoperatively.Results:Patients treated according to the pathway had shorter mean length of stay (31 vs 57 hours, P < .001), decreased cumulative morphine consumption (77 vs 727 μg/kg, P < .001), shorter time to initiate oral intake (9.3 vs 22 hours, P = .01), and greater volume of oral intake in first 24 hours postoperatively (379 vs 171 mL, P < .001). There were no differences in total anesthesia time, total surgical time, or complication rates between the control and treatment groups.Conclusions:Implementation of a standardized perioperative clinical care pathway for primary cleft palate repair is safe, feasible, and associated with reduced length of stay, reduced opioid consumption, and improved oral intake postoperatively.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-18T08:59:38Z
      DOI: 10.1177/10556656211017409
       
  • Conversion Furlow Palatoplasty and the Use of Preoperative Video
           Nasendoscopy

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      Authors: Alison Kaye, Meghan Tracy, Janelle Noel-MacDonnell, Kathryn Dent
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To assess outcomes after conversion Furlow palatoplasty with and without routine preoperative flexible fiberoptic video nasendoscopy (FFVN).Design:Retrospective cohort study.Setting:Tertiary Children’s Hospital.Patients:Greater than 3 years of age with cleft palate and velopharyngeal insufficiency (VPI) after straight-line palatoplasty requiring secondary surgery performed with a Furlow palatoplasty.Main Outcome Measures:The number of children with and without routine FFVN prior to conversion Furlow palatoplasty for VPI after initial straight-line palatoplasty. Groups were compared for surgical timing, speech outcomes, and need for additional surgery after conversion Furlow palatoplasty.Results:Fifty-eight patients underwent preoperative FFVN versus 29 without. Mean age at FFVN was 73.8 (SD 34) months. Mean age for secondary palatal surgery by conversion Furlow palatoplasty was 81.5 (SD 34.8) months with FFVN versus 73.4 (SD 34.0) months without FFVN. There was a significant difference (P < .001) for VPI diagnosis and time to surgery between the groups. Preoperative hypernasality ratings were similar between groups. Postoperatively 65.5% of FFVN and non-FFVN patients corrected to normal resonance. Only 6.9% of all patients rated moderate-severe hypernasality after surgery compared to 42.5% preoperatively. Of total, 5.7% of patients had unchanged hypernasality and only 1 patient rated worse. Seven patients ultimately required additional surgery in attempt to normalize their resonance.Conclusions:Routine preoperative FFVN does not offer any advantage for improved outcomes in children undergoing conversion Furlow palatoplasty after straight-line repair. Routine preoperative FFVN was associated with increased time to surgery after diagnosis of VPI compared to those without FFVN.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-18T08:42:51Z
      DOI: 10.1177/10556656211015008
       
  • Craniofacial Growth Analysis of Individuals With and Without Cleft Lip and
           Palate in Colombia

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      Authors: Carolina Téllez-Conti, Ingrid Isabel Mora-Diaz, David Díaz-Báez, Isomar Josefina Ocampo-Arias, Norma Edith Jiménez-Luna, Juan Camilo Niño-Paz, María Clara González-Carrera
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:Craniofacial growth is a dynamic and unpredictable process influenced by genetic and environmental factors, presenting phenotypic and gender differences.Objective:Evaluate the differences in craniofacial growth and development in a group of Colombian individuals with complete unilateral and bilateral cleft lip and palate (CLP) and without CLP, classified by gender and age.Setting and Sample Population:Five hundred forty-one profile radiographs of 126 patients with unilateral CLP, 126 with bilateral CLP, and 289 without CLP. All patients of affected groups had a history of CLP correction surgery without nasoalveolar molding with orthopedic and orthodontic treatments.Materials and Methods:This cross-sectional study was performed comparing 8 cephalometric measurements on radiographs, 5 linear/3 angular. Analysis was performed by median and interquartile range for all cephalometric measurements. Comparison between the groups was performed using Kruskal-Wallis and Mann-Whitney U, with a 95% confidence.Results:Significant differences between the groups of patients with and without CLP, between types of clefts and genders. The skeletal structures of patients with CLP were smaller than those of control but improved with growth. Patients with unilateral CLP presented flat profiles and predominant class III malocclusions, while patients with bilateral CLP, at early ages, were class II and in the prepubertal stage, the values were progressively negative until the end of the growth period, suggesting class III. Patients with CLP presented posteroinferior rotation of the mandible, vertical measurements increased, and deflection of the cranial base.Conclusion:Given their growth alterations, patients with CLP benefit from orthopedic and orthodontic treatment.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-18T08:39:09Z
      DOI: 10.1177/10556656211013690
       
  • Estimating Craniofacial Growth Cessation: Comparison of Asymptote- and
           Rate-Based Methods

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      Authors: Anna M. Hardin, Ryan P. Knigge, Hee Soo Oh, Manish Valiathan, Dana L. Duren, Kieran P. McNulty, Kevin M. Middleton, Richard J. Sherwood
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To identify differences between asymptote- and rate-based methods for estimating age and size at growth cessation in linear craniofacial measurements.Design:This is a retrospective, longitudinal study. Five linear measurements were collected from lateral cephalograms as part of the Craniofacial Growth Consortium Study (CGCS). Four estimates of growth cessation, including 2 asymptote- (GCasym, GCerr) and 2 rate-based (GCabs, GC10%) methods, from double logistic models of craniofacial growth were compared.Participants:Cephalometric data from participants in 6 historic longitudinal growth studies were included in the CGCS. At least 1749 individuals (870 females, 879 males), unaffected by craniofacial anomalies, were included in all analyses. Individuals were represented by a median of 11 images between 2.5 and 31.3 years of age.Results:GCasym consistently occurred before GCerr and GCabs consistently occurred before GC10% within the rate-based approaches. The ordering of the asymptote-based methods compared to the rate-based methods was not consistent across measurements or between males and females. Across the 5 measurements, age at growth cessation ranged from 13.56 (females, nasion-basion, GCasym) to 24.39 (males, sella-gonion, GCerr).Conclusions:Adolescent growth cessation is an important milestone for treatment planning. Based on our findings, we recommend careful consideration of specific definitions of growth cessation in both clinical and research settings since the most appropriate estimation method may differ according to patients’ needs. The different methods presented here provide useful estimates of growth cessation that can be applied to raw data and to a variety of statistical models of craniofacial growth.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-17T02:19:39Z
      DOI: 10.1177/10556656211002675
       
  • Impact of Hospital Characteristics on Mandibular Distraction Osteogenesis
           Outcomes Among Patients With Pierre Robin Sequence Utilizing a National
           Inpatient Database

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      Authors: Alexander P. Marston, Terral Patel, Phayvanh P. Pecha, Shaun A. Nguyen, Christopher M. Discolo
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:This study collected national inpatient data to investigate the impact of hospital specialty and size on patient outcomes following mandibular distraction osteogenesis (MDO).Design:Kids’ Inpatient Database was used to identify patients less than 12 months of age with Pierre Robin sequence (PRS) who underwent MDO in one of the following years: 2006, 2009, and 2012.Setting:Inpatient database from the United States.Participants:Two hundred seventy-six patients with PRS underwent MDO with 134 (48.6%) identified as nonsyndromic and 142 (51.4%) as syndromic.Interventions:Mandibular distraction osteogenesis.Main Outcome Measures:Length of hospital stay, adjunct airway and nutritional interventions and disposition.Results:The average length of stay was 24 and 30 days for patients with nonsyndromic and syndromic PRS, respectively (P = .066). Patients with a syndromic as compared to nonsyndromic diagnosis had a higher incidence of gastrostomy tube placement (21.8 vs 12.7%, P = .045). Univariate analysis showed that a lower proportion of patients at children’s hospitals as compared to non-children’s hospitals necessitated 1 or more airway or nutrition-related intervention (19/148 [12.8%] vs 31/127 [24.4%]; P = .012) and had a lower incidence of a nonroutine discharge (transfer or patient death; 7.4% vs 40.0% nonroutine; P < .001). Multivariable analysis additionally revealed that patients at children’s hospitals were less likely to discharge nonroutine (OR = 0.07, 95% CI: 0.02-0.32).Conclusions:Results from this national cohort demonstrated that at children-specific hospitals patients with PRS were less likely to require additional airway and nutritional procedures and more likely to discharge to home.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-12T09:30:20Z
      DOI: 10.1177/10556656211015007
       
  • Response to Barriers and Facilitators to the International Implementation
           of Standardized Outcome Measures in Clinical Cleft Practice

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      Authors: Conrad J. Harrison, Jeremy N. Rodrigues, Dominic Furniss, Marc C. Swan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.

      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-11T09:15:15Z
      DOI: 10.1177/10556656211015013
       
  • The Online Attention to Cleft Lip and Palate Research: An Altmetric
           Analysis

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      Authors: Serene Badran, Yazan Hassona
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:To identify research articles related to cleft lip and/or cleft palate (CL/P) that generated the highest online attention.Methods:Altmetric Explorer was used to identify the 100 articles with the highest Altmetric Attention Score (AAS). Descriptive and correlation statistics were performed to study the characteristics of these articles in relation to their publication data, research type and domain, number of Mendeley readers, and dimensions citations. Citation counts were extracted from Scopus and Google Scholar.Results:The median AAS for the top 100 outputs was 22 (range from 12 to 458). The outputs were mostly discussed on Twitter (median = 8; range = 0-131). Topics discussing treatment and care for patients with CL/P accounted for 38% of the articles with the highest AAS followed by etiology and risk factors (32%). The majority of articles originated from the USA (46%) followed by Europe (16%) and the United Kingdom (15%). No significant differences were observed in AAS among different study designs, topic domains, journals’ ranking and impact factor, and the number of citations in Scopus and Google Scholar.Conclusions:Researchers should consider use of social platforms to disseminate their work among scholars and nonscholars. Altmetrics can be combined with traditional metrics for a more comprehensive assessment of research impact.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-11T09:14:34Z
      DOI: 10.1177/10556656211014077
       
  • A Preliminary Study of Anatomical Changes Following the Use of a Pedicled
           Buccal Fat Pad Flap During Primary Palatoplasty

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      Authors: Katelyn J. Kotlarek, Michael S. Jaskolka, Xiangming Fang, Charles Ellis, Silvia S. Blemker, Bruce Horswell, Paul Kloostra, Jamie L. Perry
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:The purpose of this study was to examine the surgical impact of the pedicled buccal fat pad (BFP) flap on the levator veli palatini (LVP) muscle and surrounding velopharyngeal (VP) anatomy following primary palatoplasty using magnetic resonance imaging (MRI).Design:Observational, prospective.Setting:MRI studies were completed at 3 different facilities. All participants with BFP flap were operated on by the same surgeon.Participants:Five pediatric participants with cleft palate with or without cleft lip (CP±L) who underwent primary palatoplasty with BFP flap placement. Comparison groups consisted of 10 participants: 5 with CP±L who did not receive the BFP flap and 5 healthy controls.Interventions:All participants underwent nonsedated MRI 2 to 5 years postoperatively.Main Outcomes and Measures:Anatomical measures of the velopharynx and LVP among the 3 participant groups.Results:Median values were significantly different among groups for velar length (P = .042), effective velar length (P = .048), effective VP ratio (P = .046), LVP length (P = .021), extravelar LVP length (P = .009), and LVP origin–origin distance (P = .030). Post hoc analysis revealed a statistically significant difference between the BFP and traditional repair groups for effective VP ratio (P = .040), extravelar LVP length (P = .033), and LVP length (P = .022).Conclusions:This study provides preliminary support that the BFP flap creates a longer velum, with increased distance between the posterior hard palate and the LVP, and a larger effective VP ratio compared to traditional surgical techniques. Future research is needed to determine whether this procedure provides a more favorable mechanism for VP closure.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-11T09:13:49Z
      DOI: 10.1177/10556656211014070
       
  • Frontal Linear Scleroderma (en coup de sabre): A Case Report

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      Authors: Yo Han Oh, Soo Hyang Lee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      En coup de sabre is an unusual variant of localized scleroderma characterized by its distinct location involving the frontoparietal region of the forehead and scalp. The authors describe a rare case of en coup de sabre in a 3-year-old boy whose disease onset was at 12 months of age. This article presents the clinical manifestations of continuous changes in the lesion photographed over time. The clinical presentation, laboratory results, and radiological findings together with a brief discussion of the management of the disease are discussed.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-10T08:46:07Z
      DOI: 10.1177/10556656211013168
       
  • Cleft Lip and/or Palate in Infants Prenatally Exposed to Opioids

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      Authors: Kerry Proctor-Williams, Brenda Louw
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To determine the prevalence and odds ratios for cleft lip and/or palate (CL/P) among infants prenatally exposed to opioids with or without neonatal opioid withdrawal syndrome (NOWS).Design:This study represents an exploratory, retrospective cohort study design of newborn medical health records from 2011 to 2016.Setting:Records were drawn from a regional health system located in South Central Appalachia.Population and Study Sample:The original population yielded 3 cohorts of infants: (1) infants with opioid exposure (OE) but not requiring pharmacological intervention (OE; N = 168); (2) infants with NOWS requiring pharmacological intervention (N = 294); and (3) infants with no opioid exposure (NOE; N = 16 090), the primary comparison group.Main Outcome:Infants in the NOWS and OE groups showed significantly increased prevalence and odds ratios for CL/P when compared to those in the NOE group.Results:Prevalence rates per 1000 live births for infants with OE (35.71) and infants with NOWS (6.80) were significantly higher than those for infants with NOE (1.37). Comparison of infants with OE to the NOE group revealed significantly increased odds for CL/P, isolated cleft palate (CP), cleft lip (CL), and cleft lip and palate (CLP) (27.05, 41.81, 19.26, 19.37, respectively; all Ps < .008). The odds ratios for infants with NOWS compared to the NOE group were significantly higher for CL/P and CP (5.00 and 10.98, respectively; Ps < .03) but not for CL and CLP.Conclusion:The results provide additional evidence that prenatal OE should be considered among the critical environmental risk factors that can contribute to CL/P.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-07T09:10:58Z
      DOI: 10.1177/10556656211013687
       
  • Child Protective Services Referral in a Cleft Lip and/or Palate
           Population: Assessment of Prevalence, Indications, and Outcomes

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      Authors: Alison Kaye, Karina Shah, Sandra Lybrand, Sarah Baysinger, Meghan Tracy
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To determine the prevalence of, reasons for, and outcomes related to Child Protective Services (CPS) referral in an isolated and syndromic cleft lip/palate population.Design:Retrospective cohort study.Setting:Tertiary Children’s Hospital.Patients:Any patient
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-07T09:10:18Z
      DOI: 10.1177/10556656211013259
       
  • Readability of Online Patient Information Relating to Cleft Palate Surgery

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      Authors: Christopher V. Lavin, Evan J. Fahy, Darren B. Abbas, Michelle Griffin, Nestor M. Diaz Deleon, Daniel K. Lee, Rohit K. Khosla, Karl Bruckman, Hermann Peter Lorenz, Derrick C. Wan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:It is important for health care education materials to be easily understood by caretakers of children requiring craniofacial surgery. This study aimed to analyze the readability of Google search results as they pertain to “Cleft Palate Surgery” and “Palatoplasty.” Additionally, the study included a search from several locations globally to identify possible geographic differences.Design:Google searches of the terms “Cleft Palate Surgery” and “Palatoplasty” were performed. Additionally, searches of only “Cleft Palate Surgery” were run from several internet protocol addresses globally.Main Outcome Measures:Flesch-Kincaid Grade Level and Readability Ease, Gunning Fog Index, Simple Measure of Gobbledygook (SMOG) index, and Coleman-Liau Index.Results:Search results for “Cleft Palate Surgery” were easier to read and comprehend compared to search results for “Palatoplasty.” Mean Flesch-Kincaid Grade Level scores were 7.0 and 10.11, respectively (P = .0018). Mean Flesch-Kincaid Reading Ease scores were 61.29 and 40.71, respectively (P = .0003). Mean Gunning Fog Index scores were 8.370 and 10.34, respectively (P = .0458). Mean SMOG Index scores were 6.84 and 8.47, respectively (P = .0260). Mean Coleman-Liau Index scores were 12.95 and 15.33, respectively (P = .0281). No significant differences were found in any of the readability measures based on global location.Conclusions:Although some improvement can be made, craniofacial surgeons can be confident in the online information pertaining to cleft palate repair, regardless of where the search is performed from. The average readability of the top search results for “Cleft Palate Surgery” is around the seventh-grade reading level (US educational system) and compares favorably to other health care readability analyses.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-07T09:09:18Z
      DOI: 10.1177/10556656211013177
       
  • Three-Dimensional Printing in Cleft Care: A Systematic Review

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      Authors: Farrukh R. Virani, Evan C. Chua, Mary Roz Timbang, Tsung-yen Hsieh, Craig W. Senders
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To determine the current applications of 3-dimensional (3D) printing in the care of patients with cleft lip and palate. We also reviewed 3D printing limitations, financial analysis, and future implications.Design:Retrospective systematic review.Methods:Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines were used by 3 independent reviewers. Articles were identified from Cochrane library, Ovid Medline, and Embase. Search terms included 3D printing, 3 dimensional printing, additive manufacturing, rapid prototyping, cleft lip, and cleft palate. Exclusion criteria included articles not in English, animal studies, reviews without original data, oral presentations, abstracts, opinion pieces, and articles without relevance to 3D printing or cleft lip and palate.Main Outcome Measures:Primary outcome measure was the purpose of 3D printing in the care of patients with cleft lip and palate. Secondary outcome measures were cost analysis and clinical outcomes.Results:Eight-four articles were identified, and 39 met inclusion/exclusion criteria. Eleven studies used 3D printing models for nasoalveolar molding. Patient-specific implants were developed via 3D printing in 6 articles. Surgical planning was conducted via 3D printing in 8 studies. Eight articles utilized 3D printing for anatomic models/educational purposes. 3-Dimensional printed models were used for surgical simulation/training in 6 articles. Bioprinting was utilized in 4 studies. Secondary outcome of cost was addressed in 8 articles.Conclusion:3-Dimensional printing for the care of patients with cleft lip and palate has several applications. Potential advantages of utilizing this technology are demonstrated; however, literature is largely descriptive in nature with few clinical outcome measures. Future direction should be aimed at standardized reporting to include clinical outcomes, cost, material, printing method, and results.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-07T09:08:41Z
      DOI: 10.1177/10556656211013175
       
  • Does the Timing of 1-Stage Palatoplasty With Radical Muscle Dissection
           Effect Long-Term Midface Growth' A Single-Center Retrospective
           Analysis

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      Authors: Vikram Shetty, Nanda Kishore Patteta, Anirudh Yadav, Devyani Bahl, Hermann F. Sailer
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate the long-term effect of timing of 1-stage palatoplasty on midfacial growth in patients with cleft lip and palate (CLP).Design:Retrospective observational cohort study.Study Setting:Institutional hospital.Patients:One hundred twelve patients with CLP who underwent palatoplasty and were divided into 3 groups: group I: operated between 9 and 11 months; group II: operated between 18 and 20 months; and group III: operated between 21 and 24 months.Interventions:All patients underwent von Langenbeck palatoplasty technique, which was converted to a Bardach 2-flap technique in case of any technical difficulties. The patients were followed up between 8 and 9 years when they reported for secondary alveolar bone grafting. Postsurgical cephalometric and dental casts measurements were taken for midfacial growth analysis.Main Outcome Measures:The cephalometric measures were analyzed for midfacial growth and compared within the groups.Results:Statistically significant difference (P < .01) was found on comparing the cephalometric parameters such as sella–nasion–A point angle (SNA), A point–nasion–B point angle (ANB), n toperpendicular to point A (N-perpA), condylon to point A (Co-A), anterior nasal spine to posterior nasal spine (ANS-PNS), nasion to Anterior nasal spine (N-ANS), nasion to menton (N Me), and witts appraisal (Witt (AO-BO)) in group I when compared to both group II and group III patients, implying deficient midfacial growth in group I. No statistical difference was found in the cephalometric values between group II and group III. Group II had better cephalometric measurements than group III, showing better growth in group II than group III. Overall, there was less incidence of midfacial hypoplasia in patients treated between 18 and 20 months (group II).Conclusion:We conclude that palatal closure carried out at 18 to 20 months and 21 to 24 months is associated with better midfacial growth when compared to closure at 9 to 11 months. The best time to operate would be between 18 and 20 months to avoid speech disturbances. Midfacial growth can be greatly influenced by the timing of 1-stage palatoplasty.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-06T09:30:51Z
      DOI: 10.1177/10556656211013174
       
  • Prevalence of Reading Difficulties in 9- to 10-Year Old Children in Sweden
           Born With Cleft Palate

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      Authors: Justin Parry Weinfeld, Jakob Åsberg Johnels, Christina Persson
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To investigate the prevalence of reading difficulties in children born with cleft palate at ages 9 and 10 in Sweden.Design:Using a cross-sectional design, a parental questionnaire assessing dyslexia-like reading difficulties (Short Dyslexia Scale, SDS) was administered together with separate questions regarding background data.Participants:Families with a child born with overt cleft palate with or without cleft lip in 4 regions of Sweden. A total of 245 families were approached of which 138 families responded. Data from 136 (56%) were complete with information on cleft type and could be analyzed.Results:Twenty-two percent (95% CI, 15-30) of the whole study group displayed risk for dyslexic reading difficulties on the SDS corresponding to the 7th to 10th percentiles in the population. Children with cleft palate only had a significantly higher prevalence of reading difficulties (37%) compared to children with unilateral cleft palate (19%) and bilateral cleft palate (10%). The frequency of reading difficulties in participants with comorbidity was 32%. Among a subgroup with reported comorbidity in areas of attention, language, and learning problems, there was a 2.5 times higher risk of reading disability compared to participants without this reported comorbidity.Conclusion:The prevalence of reading difficulties in the cleft palate population was higher than in the general population. Results showed that co-occurring difficulties were common in the cleft group and that reading difficulties often appear together with other, co-occurring neurodevelopmental difficulties.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-06T09:30:37Z
      DOI: 10.1177/10556656211013236
       
  • Safety and Efficacy of Single-Dose Ketorolac for Postoperative Pain
           Management After Primary Palatoplasty: A Prospective Cohort Study With
           Historical Controls

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      Authors: Jason R. Stein, Esperanza Mantilla-Rivas, Marudeen Aivaz, Md Sohel Rana, Ishwarya Shradha Mamidi, Brynne A. Ichiuji, Monica Manrique, Gary F. Rogers, Julia C. Finkel, Albert K. Oh
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To analyze safety and efficacy of single-dose ketorolac after primary palatoplasty (PP).Design:Consecutive cohort of patients undergoing PP, comparing to historical controls. Setting: A large academic children’s hospital.Patients, Participants:A consecutive cohort of 111 patients undergoing PP (study n = 47) compared to historical controls (n = 64).Interventions:All patients received intraoperative acetaminophen, dexmedetomidine, and opioids while the study group received an additional single dose of ketorolac (0.5 mg/kg) at the conclusion of PP.Main Outcome Measures:Safety of ketorolac was measured by significant bleeding complications and need for supplementary oxygen. Efficacy was assessed through bleeding, Face Legs Activity Cry Consolability (FLACC) scale, and opioid dose.Results:Length of stay was similar for both groups (control group 38.5 hours [95% CI: 3.6-43.3] versus study group 37.6 hours [95% CI: 31.3-44.0], P = .84). There were no significant differences in all postoperative FLACC scales. The mean dose of opioid rescue medication measured as morphine milligram equivalents did not differ between groups (P = .56). Significant postoperative hemorrhage was not observed.Conclusions:This is the first prospective study to evaluate the safety and efficacy of single-dose ketorolac after PP. Although lack of standardization between study and historical control groups may have precluded observation of an analgesic benefit, analysis demonstrated a single dose of ketorolac after PP is safe. Further investigations with more patients and different postoperative regimens may clarify the role of ketorolac in improving pain after PP.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-05-04T09:44:32Z
      DOI: 10.1177/10556656211012864
       
  • Dental Arch Relationship Outcomes Following 2-Stage Palatoplasty for
           Japanese Patients With Complete Unilateral Cleft Lip and Palate: A
           3-Center Study

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      Authors: Junya Kato, Tadashi Mikoya, Yumi Ito, Yoshiaki Sato, Setsuko Uematsu, Yasumitsu Kodama, Takafumi Susami, Tadashi Yamanishi, Ritsuo Takagi
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To compare dental arch relationship outcomes following 3 different 2-stage palatal repair protocols.Design:Retrospective, cross sectional.Setting:Three cleft palate centers (A, B, C) in Japan.Patients:Ninety (A: 39, B: 26, C: 25) consecutively treated Japanese patients with complete unilateral cleft lip and palate.Interventions:In A, the soft palate and the posterior half of the hard palate were repaired at a mean age of 1 year 7 months. In B, the soft palate and hard palate were closed separately at a mean age of 1 year 6 months and 5 years 8 months, respectively. In C, the soft palate and hard palate were closed at a mean age of 1 year and 1 year 5 months, respectively.Main Outcome Measures:Dental arch relationships were assessed using the 5-Year-Olds’ (5-Y) index by 5 raters and the Huddart/Bodenham (HB) index by 2 raters.Results:Intra- and inter-rater reliabilities showed substantial or almost perfect agreement for the 5-Y and HB ratings. No significant differences in mean values and distributions of 5-Y scores were found among the 3 centers. The mean HB index scores of molars on the minor segment were significantly smaller in C than those in A and B (P < .05).Conclusions:There were no significant differences in dental arch relationships at 5 years among the times and techniques of hard palate closure. However, further analysis of the possible influence of infant cleft size as a covariable on a larger sample size is needed.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-29T07:02:38Z
      DOI: 10.1177/10556656211010606
       
  • Cleft Lip and Palate in Infants With Prenatal Opioid Exposure

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      Authors: Olivia Langa, Alex T. Cappitelli, Ingrid M. Ganske
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:This study examines phenotypic presentation and perioperative outcomes of cleft-related procedures for infants with cleft lip and/or palate (CL/P) and prenatal opioid exposure.Design:This is a retrospective review of infants with prenatal opioid exposure treated for CL/P from 2008 to 2018.Setting:Patients cared for at a tertiary center from 2008 to 2018.Patients/Participants:Eighteen patients with documented prenatal opioid exposure and CL/P had primary repairs in our unit.Main Outcome Measure(s):The phenotypes of CL/P were characterized. Demographic data regarding additional exposures, as well as associated medical and social comorbidities were recorded. Outcome variables included operative delays, perioperative complications, and loss of follow-up.Results:Isolated cleft palate (CP; 67%) was overrepresented among patients with prenatal opioid exposure and CL/P, as was Robin sequence (50% in isolated CP). Fifty-six percent had exposure to additional substances. A majority (67%) had other medical conditions or anomalies, and 17% had known genetic syndromes. Seventy-two percent were in state custody. Thirty-nine percent of exposed patients had delays in their planned operative dates due to medical and/or social factors. There were no postoperative readmissions following cleft procedures. Lack of follow-up was noted in 33% of patients.Conclusions:Infants with CL/P who have prenatal opioid exposure are likely to have additional medical conditions and complex social challenges.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-28T07:42:03Z
      DOI: 10.1177/10556656211011896
       
  • A Novel IRF6 Frameshift Mutation in a Large Chinese Pedigree With Van der
           Woude syndrome

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      Authors: Qi Peng, Wenyan Qin, Siping Li, Meihua Huang, Chunbao Rao, Xiaomei Lu
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Aims:Van der Woude syndrome (VWS) is one of the most common craniofacial anomalies, causing significant functional and psychological burden to the patients. This study aimed to identify the genetic cause of VWS in a Chinese family.Methods:Whole genome sequencing (WGS) was performed to screen for pathogenic mutations. Various Bioinformatics tools were used to assess the pathogenicity of the variants. Cosegregation analysis of the candidate variant was carried out. Interpretation of variants was performed according to the American College of Medical Genetics and Genomics guidelines.Results:A novel frameshift duplication c.373_374dupAA (p.Asn125Lys fs*43) was identified in exon 4 of the interferon regulatory factor 6 (IRF6) gene in all 3 affected members, which were not found in unaffected family members. The novel mutation leads to a frameshift and a premature stop codon which caused putative truncated protein. Protein alignment indicated high evolutionary conservation of the p.N125 residue, and this mutation was predicted by online tools to be damaging and deleterious.Conclusions:This study demonstrates that the novel mutation c.373_374dupAA (p.Asn125Lysfs*43) in the IRF6 gene corresponds to the VWS in this family. The discovery of this pathogenic variant enriches the genotypic spectrum of IRF6 gene and contributes to genetic diagnosis and counseling of families with VWS.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-28T07:28:24Z
      DOI: 10.1177/10556656211010909
       
  • Birth Defect Co-Occurrence Patterns Among Infants With Cleft Lip and/or
           Palate

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      Authors: Maria Luisa Navarro Sanchez, Renata H. Benjamin, Laura E. Mitchell, Peter H. Langlois, Mark A. Canfield, Michael D. Swartz, Angela E. Scheuerle, Daryl A. Scott, Hope Northrup, Christian P. Schaaf, Joseph W. Ray, Scott D. McLean, Han Chen, Philip J. Lupo, A.J. Agopian
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To investigate 2- to 5-way patterns of defects co-occurring with orofacial clefts using data from a population-based registry.Design:We used data from the Texas Birth Defects Registry for deliveries between 1999 and 2014 to Texas residents, including 1884 cases with cleft palate (CP) and 5289 cases with cleft lip with or without cleft palate (CL±P) without a known syndrome. We identified patterns of defects co-occurring with CP and with CL±P observed more frequently than would be expected if these defects occurred independently. We calculated adjusted observed-to-expected (O/E) ratios to account for the known tendency of birth defects to cluster nonspecifically.Results:Among infants without a syndrome, 23% with CP and 21% with CL±P had at least 1 additional congenital anomaly. Several combinations of defects were observed much more often than expected. For example, the combination of CL±P, congenital hydrocephaly, anophthalmia, and other nose anomalies had an O/E ratio of 605. For both CP and CL±P, co-occurrence patterns with the highest O/E ratios involved craniofacial and brain abnormalities, and many included the skeletal, cardiovascular, and renal systems.Conclusions:The patterns of defects we observed co-occurring with clefts more often than expected may help improve our understanding of the relationships between multiple defects. Further work to better understand some of the top defect combinations could reveal new phenotypic subgroups and increase our knowledge of the developmental mechanisms that underlie the respective defects.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-28T07:27:44Z
      DOI: 10.1177/10556656211010060
       
  • The Relationship Between Maxillary Growth and Speech in Children With a
           Unilateral Cleft Lip and Palate at 5 Years of Age

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      Authors: Matthew Fell, Jibby Medina, Kate Fitzsimons, Miriam Seifert, Anne Roberts, Craig Russell, Scott Deacon
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:This study sought to investigate the association between maxillary growth and speech outcomes for children with a repaired unilateral cleft lip and palate (UCLP) at 5 years of age.Participants:In all, 521 children (180 females and 341 males) with a nonsyndromic complete UCLP, born between 2007 and 2012 in England, Wales, and Northern Ireland were included in this study.Outcome Measures:Maxillary growth was analyzed using dental models scored by the 5-Year-Olds’ index, and perceptual speech analyses were scored by the Cleft Audit Protocol for Speech – Augmented rating.Results:Forty-one percent of the children achieved good maxillary growth (scores 1 and 2 on 5-Year-Old’ index). Fifty percent of the children achieved normal speech (achieving UK speech standard 1). Maxillary growth was not found to have an impact on speech outcome when described by the 3 UK National Cleft Lip and Palate Speech Audit Outcome Standards. Analysis according to individual speech parameters showed dentalizations to be less prevalent in children with good maxillary growth compared to fair and poor growth (P = .001). The remaining speech parameters within resonance, nasal airflow, and articulation categories were not significantly associated with maxillary growth.Conclusion:The findings from this study suggest that children with a history of complete UCLP, who have poor maxillary growth, are not at a higher risk of having major speech errors compared to children with good or fair maxillary growth at 5 years of age.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-23T07:12:19Z
      DOI: 10.1177/10556656211010620
       
  • The Performance of Secondary Nasal Alar Base Revision for Unilateral Cleft
           

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      Authors: Yoshitaka Matsuura, Hideaki Kishimoto
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Although primary surgery for cleft lip has improved over time, the degree of secondary cleft or nasal deformity reportedly varies from a minimum degree to a remarkable degree. Patients with cleft often worry about residual nose deformity, such as a displaced columella, a broad nasal floor, and a deviation of the alar base on the cleft side. Some of the factors that occur in association with secondary cleft or nasal deformity include a deviation of the anterior nasal spine, a deflected septum, a deficiency of the orbicularis muscle, and a lack of bone underlying the nose. Secondary cleft and nasal deformity can result from incomplete muscle repair at the primary cleft operation. Therefore, surgeons should manage patients individually and deal with various deformities by performing appropriate surgery on a case-by-case basis. In this report, we applied the simple method of single VY-plasty on the nasal floor to a patient with unilateral cleft to revise the alar base on the cleft side. We adopted this approach to achieve overcorrection on the cleft side during surgery, which helped maintain the appropriate position of the alar base and ultimately balanced the nose foramen at 13 months after the operation. It was also possible to complement the height of the nasal floor without a bone graft. We believe that this approach will prove useful for managing cases with a broad and low nasal floor, thereby enabling the reconstruction of a well-balanced nose.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-23T07:11:21Z
      DOI: 10.1177/10556656211010609
       
  • A Simple Method for Eye Protection During Prone Positioning for
           Craniofacial Surgery

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      Authors: Jaime Grant, Peter Noons, Edmund D. Carver, Martin J. Evans, Nicholas White, M. Stephen Dover, Desiderio Rodrigues
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Surgery in the prone position risks vision loss due to a number of factors. Craniofacial surgery poses an even greater risk due to the anatomical and physiological makeup of these patients. Here, we describe a novel method of providing protection from direct pressure on the globe during prone positioning for craniofacial procedures and our protocol for improving safety and reducing the risk of postoperative vision loss.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-22T07:32:25Z
      DOI: 10.1177/10556656211010603
       
  • Long-Term Effects of Nasoalveolar Molding in Patients With Unilateral
           Cleft Lip and Palate: A Systematic Review and Meta-Analysis

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      Authors: William M. Padovano, Gary B. Skolnick, Sybill D. Naidoo, Alison K. Snyder-Warwick, Kamlesh B. Patel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:The aim of this systematic review and meta-analysis was to investigate long-term treatment effects of nasoalveolar molding (NAM) in patients with unilateral cleft lip and palate (UCLP).Design:Included manuscripts met the following criteria: (1) involved patients with UCLP who received NAM; (2) included comparison group(s) who either received non-NAM passive presurgical infant orthopedic appliances (PSIO) or who did not receive any PSIO; (3) reported at least one objective or validated measure of nasolabial, craniofacial, or palatal form; and (4) had patient follow-up beyond 4 years of age.Results:A total of 12 studies were included in this review. Meta-analyses were possible for Asher-McDade parameters and cephalometric measurements. Compared to patients who did not receive any PSIO, those who underwent NAM therapy were more likely to have good to excellent frontal nasal form (Risk ratio: 2.4, 95% CI: 1.24-3.68) and vermillion border (Risk ratio: 1.8, 95% CI: 1.19-2.71). However, there were no statistically significant differences in cephalometric measurements between these groups. Additionally, there were no statistically significant differences between patients receiving NAM versus non-NAM PSIO. There was insufficient evidence to determine the impact of NAM on dental arch development.Conclusions:The preponderance of evidence in this review suggests that NAM produces benefits in nasolabial aesthetic form when compared with no appliance-based presurgical treatment. However, there is insufficient evidence to conclude whether NAM produces such benefits when compared with other passive PSIOs.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-22T07:31:05Z
      DOI: 10.1177/10556656211009702
       
  • The Palatal Fistula Closure Using Buccal Fat Graft After Palatoplasty for
           Cleft Palate: Two Case Reports

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      Authors: Kiichiro Yaguchi, Kenya Fujita, Masahiko Noguchi, Fumio Nagai, Shunsuke Yuzuriha
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Fistula recurrence is high after secondary follow-up operation to close the fistula after primary palatal surgery. Therefore, preventing fistula recurrence is important. Here, we describe the technique of closing palatal fistula after palatal surgery with a buccal fat graft in 2 cases. We elevate the mucosal flap around the palatal fistula, suture the nasal mucosa, transplant the buccal fat between the nasal and oral mucosa for the palatal fistula after palatal surgery, and suture the oral mucosa. Palatal fistula did not recur after surgery. This method is simple and useful for suturable fistula and does not require a local flap.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-22T07:30:17Z
      DOI: 10.1177/10556656211007000
       
  • Three-Dimensional Dynamic Analysis of the Reproducibility of Verbal and
           Nonverbal Facial Expressions

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      Authors: Tiancheng Qiu, Zulin Xue, Ling Wu, Rong Yang, Xiaojing Liu, Xing Wang, Zili Li
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:The aim of this study is to compare the short- and long-term reproducibility of verbal and nonverbal facial expressions of normal people using dynamic 3-dimensional (3-D) imaging.Design:Prospective, cross-sectional, controlled study.Setting:Peking University School and Hospital of Stomatology, Beijing, China.Patients and Participants:Twenty-seven participants, 12 males and 15 females, were recruited for this study.Methods:A 3-D dynamic system was applied to capture the process of 4 nonverbal facial expressions (smile lips closed, smile lips open, lip purse, cheek puff) and 2 verbal facial expressions (/i:/, /u:/) at an initial time point, 15 minutes later, and 1 week later. Key frames were selected from each expression recording sequence.Main Outcome Measures:The root mean square (RMS) between each key frame and its corresponding frame at rest was calculated. ΔRMS reflected the difference of the same key frames between the different sessions of the same expression of the same participant. The reproducibility of different facial expressions at different time intervals were analyzed.Results:There was no significant difference in verbal and nonverbal expression repeatability during a 15-minute interval, except for cheek puff motion. Following a 1-week interval, verbal expression repeatability was superior to that of nonverbal expressions (P < .01). Compared with nonverbal expressions, the repeatability of verbal expressions did not obviously decrease with the increase in recording interval.Conclusions:Dynamic 3-D imaging is a useful technique for facial expression analysis. Verbal expressions showed greater reproducibility than nonverbal expressions.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-22T07:23:06Z
      DOI: 10.1177/10556656211001998
       
  • Longitudinal Follow-Up of Facial Growth of Patients With Unilateral Cleft
           Lip and Palate Following Modified Veau–Wardill–Kilner Palatoplasty

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      Authors: Konomi Iwasaki, Yuki Sato Yamamoto, Marina Masuda, Prasad Nalabothu, Fumio Ohkubo, Koichi Kadomatsu, Koutaro Maki
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate facial growth after modified Veau–Wardill–Kilner/pushback (PB) palatoplasty from childhood to adulthood in patients with unilateral cleft lip and palate (UCLP).Design:Retrospective longitudinal study.Setting:Single center.Patients:Sixty-two (29 males and 33 females) consecutive patients with nonsyndromic UCLP.Interventions:Pushback palatoplasty and subsequent cleft palate treatment.Main Outcome Measures:Lateral and posterior–anterior cephalograms were taken at 3 different phases: phase 1 (before first-stage orthodontic treatment; N = 58; average age, 4.9 ± 1.1 years), phase 2 (before second-stage orthodontic treatment; N = 58; 15.9 ± 1.1 years), and phase 3 (after orthodontic retention; N = 51; 22.1 ± 3.2 years).Results:The majority of patients had skeletal class III morphology in all 3 phases due to retrognathic maxilla. Maxillary growth did not improve in phase 2 despite first-stage orthodontic treatment in phase 1. Maxillary morphology improved in phase 3 but retardation occurred, although 77.42% of patients received orthognathic surgery during second-stage orthodontic treatment. Mandibular growth was slightly reduced in phases 1 and 2 and the mandible remained retrognathic in phase 3, following mandibular setback orthognathic surgery. The horizontal occlusal cant was slightly upward and toward the cleft side with respect to the reference plane, and the upper midline was deviated to the cleft side in phases 1 and 3.Conclusions:Patients with UCLP who undergo phased PB palatoplasty, orthodontic treatment, and orthognathic surgeries do not maintain skeletal class I facial morphology.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-15T08:00:08Z
      DOI: 10.1177/10556656211004852
       
  • Orthodontic Pre Grafting Closure of Large Alveolar Bony and Soft Tissue
           Gaps: A Novel Nonsurgical Protraction of the Lesser Segments in Growing
           Patients With Cleft Lip and Palate

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      Authors: Maria Costanza Meazzini, Noah Cohen, Valeria Marinella Augusta Battista, Cristina Incorvati, Federico Biglioli, Luca Autelitano
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:Closure of wide alveolar clefts with large soft tissue gaps and reconstruction of the dentoalveolar defect are challenging for the surgeon. Some authors successfully used interdental segmental distraction, which requires an additional surgical procedure.Objective:This study evaluates the effectiveness of tooth borne devices utilized to orthopedically advance the lesser segments, with a complete approximation of the soft tissue of the alveolar stumps, allowing traditional simultaneous soft tissue closure and bone grafting, and avoiding the need for supplementary surgery.Methods:Eight growing patients, 2 with unilateral complete cleft lip and palate (UCLP) and 6 with bilateral complete cleft lip and palate (BCLP), with large soft tissue and bony alveolar defects prior to bone grafting were prospectively selected. A banded rapid palatal expander (RPE) in BCLP and a modified RPE in UCLP combined with protraction face mask in younger patients or a modified Alt-Ramec in patients older than 12 years were applied. Radiographic and photographic records were available at T0, at the end of protraction (T1) and at least 1 year after bone grafting (T2).Results:Patients with large gaps showed a significant reduction in the bony cleft area and approximation of the soft tissues at T1. All patients received bone grafting with good healing and ossification at T2.Conclusion:In growing patients with UCLP and BCLP with large gaps, presurgical orthodontic protraction seems to be an efficient method to reduce the cleft defect, minimizing the risk of post grafting fistulas, reducing the need for supplementary surgical procedures.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-13T07:37:00Z
      DOI: 10.1177/10556656211007697
       
  • Nonsurgical Orthodontic Airway Plate Treatment for Newborns With Robin
           Sequence

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      Authors: HyeRan Choo, Rohit K. Khosla, Kara D. Meister, Derrick C. Wan, Hung-Fu C. Lin, Robert Feczko, Karl Bruckman, Elena Hopkins, Mai Thy Truong, Hermann P. Lorenz
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Despite promising outcomes for >50 years, nonsurgical orthodontic airway plates (OAP) are only infrequently offered for babies with Robin sequence in a few parts of the world. This article demonstrates possibility of providing functional improvement using an OAP to help these babies overcome their functional and structural difficulties on their own. Two consecutively treated cases are presented exemplifying that OAP treatment that had originated from Europe is reproducible and effective in an institution in the United States.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-13T07:36:00Z
      DOI: 10.1177/10556656211007689
       
  • Skeletal Maturation in Patients With Cleft Lip and/or Palate: A Systematic
           Review

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      Authors: Xiaoyi Wu, Wenying Kuang, Jie Zheng, Zhengkun Yang, Meiqing Ren, Ruicong Yang, Wenjun Yuan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:The objective of this systematic review was to evaluate the evidence regarding skeletal maturation in patients with cleft lip and/or palate (CL/P) and to investigate whether the skeletal maturation is delayed in these patients.Design:Systematic review.Methods:Electronic and manual searches of scientific literature were conducted in 4 databases (MEDLINE, Embase, Cochrane Library, and Web of Science). Cohort studies that compared the skeletal maturation of patients with CL/P with that of children without CL/P were eligible for inclusion. The quality of included cohort studies was assessed using the Newcastle-Ottawa Scale.Patients and Participants:Patients of any sex and ethnicity with CL/P and children without CL/P were included in this systematic review.Main Outcome Measures:Difference in skeletal maturation between patients with CL/P and patients without CL/P.Results:Thirteen retrospective cohort studies were included in this systematic review. Ten studies were considered of high quality and 3 were considered of general quality. The results of the included studies comparing skeletal maturation of patients with CL/P and children without CL/P were heterogeneous.Conclusion:Heterogeneity of skeletal maturation assessment methods, chronological age, sex, cleft type, and race may influence the final results of clinical studies on skeletal maturation in patients with CL/P. Overall, there is limited evidence to determine whether the skeletal maturation level of patients with CL/P is delayed compared to that of normal children. Further studies are needed to determine the skeletal maturation patterns in patients with CL/P.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-08T08:24:18Z
      DOI: 10.1177/10556656211007680
       
  • Cleft Alveolar Bone Graft Materials: Literature Review

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      Authors: Caroline Dissaux, Laetitia Ruffenach, Catherine Bruant-Rodier, Daniel George, Frédéric Bodin, Yves Rémond
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:Since the early stages of alveolar bone grafting development, multiple types of materials have been used. Iliac cancellous bone graft (ICBG) remains the gold standard.Design/Methods:A review of literature is conducted in order to describe the different bone filling possibilities, autologous or not, and to assess their effectiveness compared to ICBG. This review focused on studies reporting volumetric assessment of the alveolar cleft graft result (by computed tomography scan or cone beam computed tomography).Results:Grafting materials fall into 3 types: autologous bone grafts, ICBG supplementary material, and bone substitutes. Among autologous materials, no study showed the superiority of any other bone origin over iliac cancellous bone. Yet ICBG gives inconsistent results and presents donor site morbidity. Concerning supplementary material, only 3 studies could show a benefit of adding platelet-rich fibrin (1 study) or platelet-rich plasma (2 studies) to ICBG, which remains controversial in most studies. There is a lack of 3-dimensional (3D) assessment in most articles concerning the use of scaffolds. Only one study showed graft improvement when adding acellular dermal matrix to ICBG. Looking at bone substitutes highlights failures among bioceramics alone, side-effects with bone morphogenetic protein-2 composite materials, and difficulties in cell therapy setup. Studies assessing cell therapy–based substitutes show comparable efficacy with ICBG but remain too few.Conclusion:This review highlights the lack of 3D assessments in the alveolar bone graft materials field. Nothing dethroned ICBG from its position as the gold standard treatment at this time.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-07T08:18:42Z
      DOI: 10.1177/10556656211007692
       
  • Reduced Socioeconomic Disparities in Cleft Care After Implementing a Cleft
           Nurse Navigator Program

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      Authors: Connor Wagner, Carrie E. Zimmerman, Carlos Barrero, Christopher L. Kalmar, Paris Butler, James Guevara, Scott P. Bartlett, Jesse A. Taylor, Nancy Folsom, Jordan W. Swanson
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate the impact of a Cleft Nurse Navigator (CNN) program on care for patients with cleft lip and cleft palate and assess the programs efficacy to reduce existing socioeconomic disparities in care.Design:Retrospective review and outcomes analysis (n = 739).Setting:Academic tertiary care center.Patients:All patients presenting with cleft lip and/or cleft palate (CL/P) born between May 2009 and November 2019 with exclusions for atypical clefts, submucous cleft palates, international adoption, and very late presentation (after 250 days of life).Interventions:Multidisciplinary care coordination program facilitated by the CNN.Main Outcome Measures:Patient age at first outpatient appointment and age at surgery, reported feeding issues, weight gain, and patient-cleft team communications.Results:After CNN implementation, median age at outpatient appointment decreased from 20 to 16 days (P = .021), volume of patient-cleft team communications increased from 1.5 to 2.8 (P < .001), and frequency of reported feeding concerns decreased (50% to 35%; P < .001). In the pre-CNN cohort, nonwhite and publicly insured patients experienced delays in first outpatient appointment (P < .001), cleft lip repair (P < .011), and cleft palate repair (P < .019) compared to white and privately insured patients, respectively. In the post-CNN cohort, there were no significant differences in first appointment timing by race nor surgical timing on the basis of racial identity nor insurance type.Conclusions:A variety of factors lead to delays in cleft care for marginalized patient populations. These findings suggest that a CNN can reduce disparities of access and communication and improve early feeding in at-risk cohorts.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-07T08:14:45Z
      DOI: 10.1177/10556656211005646
       
  • One-Year Treatment Outcome of Profile Changes After Transcutaneous
           Maxillary Distraction Osteogenesis in Growing Children With Cleft Lip and
           Palate

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      Authors: Wei-Ling Gao, Yi-Hao Lee, Chi-Yu Tsai, Te-Ju Wu, Jui-Pin Lai, Shiu-Shiung Lin, Yu-Jen Chang
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate the long-term stability of LeFort I osteotomy followed by distraction osteogenesis with a transcutaneous rigid external device for the treatment of severe maxillary hypoplasia in patients with cleft lip and palate.Patients and Methods:Nine patients with cleft lip and palate underwent rigid external distraction after a LeFort I osteotomy for maxillary advancement. Lateral cephalometric films were analyzed for assessment of treatment outcome and stability in 1 month, 6 months, and 1 year after distraction.Results:Significant maxillary advancement was observed in the horizontal direction, with the anterior nasal spine (ANS) distance of the maxilla increasing by an average of 20.5 ± 5.1 mm after distraction. The ANS relapse rates in 6 months and 1 year were 8.7% and 12.8%, respectively. The mean inclination of upper incisors to the palatal plane was almost unchanged (before: 109.8° ± 6.6°; after: 108.9° ± 7.5°). The movement ratios at the nasal tip/ANS, soft tissue A point/A point, and the upper vermilion border/upper incisor edge were 0.36:1, 0.72:1, and 0.83:1, respectively.Conclusion:Considerable maxillary advancement was achieved with less change of incisors inclination after distraction. Moreover, the relapse rate after 1 year was minimal. The concave facial profile was improved as well as the facial balance and aesthetics.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-05T07:36:55Z
      DOI: 10.1177/10556656211005638
       
  • Cultural Beliefs on the Causes of Cleft Lip and/or Palate in Malaysia: A
           Multicenter Study

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      Authors: Ilyasak Hussin, Ahmad Sukari Halim, Mohd Ismail Ibrahim, Zara Octavia Markos, Eryana Susantri A. Bakar Effendie
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To identify the cultural beliefs about the causes of cleft among parents of patients with nonsyndromic cleft lip and/or palate in a multiethnic society in Malaysia and the difficulties encountered in receiving cleft treatment.Design:A descriptive cross-sectional multicenter study based on a study questionnaire was conducted of parents of patients with cleft lip and/or palate.Setting:Three centers providing cleft care from different regions in Malaysia: the national capital of Kuala Lumpur, east coast of peninsular Malaysia, and East Malaysia on the island of Borneo.Participants:Parents/primary caregivers of patients with cleft lip and/or palate.Results:There were 295 respondents from different ethnic groups: Malays (58.3%), indigenous Sabah (30.5%), Chinese (7.1%), Indian (2.4%), and indigenous Peninsular Malaysia and Sarawak (1.7%). Malay participants reported that attributing causes of cleft to God’s will, superstitious beliefs that the child’s father went fishing when the mother was pregnant or inheritance. Sabahans parents reported that clefts are caused by maternal antenatal trauma, fruit picking, or carpentry. The Chinese attribute clefts to cleaning house drains, sewing, or using scissors. Cultural background was reported by 98.3% of participants to pose no barrier in cleft treatment. Those from lower socioeconomic and educational backgrounds were more likely to encounter difficulties while receiving treatment, which included financial constraints and transportation barriers.Conclusion:There is a wide range of cultural beliefs in the multiethnic society of Malaysia. These beliefs do not prevent treatment for children with cleft. However, they face challenges while receiving cleft treatment, particularly financial constraints and transportation barriers. Such barriers are more likely experienced by parents from lower income and lower education backgrounds.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-05T07:32:37Z
      DOI: 10.1177/10556656211003797
       
  • Does Sleep Position Influence Sleep-Disordered Breathing in Infants With
           Cleft Palate: A Feasibility Study'

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      Authors: Clare S. Murray, Tanya Walsh, Trisha Bannister, Aleksandra Metryka, Karen Davies, Yin Ling Lin, Paula Williamson, Peter Callery, Kevin O’Brien, William Shaw, Iain Bruce
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Cleft palate (CP) can affect breathing, leading to sleep-disordered breathing (SDB). Sleep position can affect SDB, but the optimum sleep position for infants with CP is unknown. We aimed to determine the design of a pragmatic study to investigate the effect of the 2 routinely advised sleep positions in infants with CP on oxygen saturations.Design:A multicentered observational cohort.Setting:Four UK-based cleft centers, 2 advising supine- and 2 side-lying sleep positions for infants with CP.Participants:Infants with isolated CP born July 1, 2015, and December 31, 2016. Of 48 eligible infants, 30 consented (17 side-lying; 13 supine).Interventions:Oxygen saturation (SpO2) and end-tidal carbon dioxide (ETCO2) home monitoring at age 1 and 3 months. Qualitative interviews of parents.Outcome Measures:Willingness to participate, recruitment, retention, and acceptability/success (>90 minutes recording) of SpO2 and ETCO2 monitoring.Results:SpO2 recordings were obtained during 50 sleep sessions on 24 babies (13 side-lying) at 1 month (34 sessions >90 minutes) and 50 sessions on 19 babies (10 side-lying) at 3 months (27 sessions >90 minutes). The ETCO2 monitoring was only achieved in 12 sessions at 1 month and 6 at 3 months; only 1 was >90 minutes long. The ETCO2 monitoring was reported by the majority as unacceptable. Parents consistently reported the topic of sleep position in CP to be of importance.Conclusions:This study has demonstrated that it is feasible to perform domiciliary oxygen saturation studies in a research setting and has suggested that there may be a difference in the effects of sleep position that requires further investigation. We propose a study with randomization is indicated, comparing side-lying with supine-lying sleep position, representing an important step toward better understanding of SDB in infants with CP.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-01T09:03:27Z
      DOI: 10.1177/10556656211003459
       
  • Mind the Gap: Alveolar Bone Graft Revision in the Era of Computed
           Tomography

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      Authors: Ema Zubovic, Gary B. Skolnick, Abdullah M. Said, Richard J. Nissen, Alison K. Snyder-Warwick, Kamlesh B. Patel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To determine the rate of revision alveolar bone grafting (ABG) in patients with cleft lip and palate (CLP) before and after the introduction of postoperative computed tomography (CT).Design:Retrospective case–control study analyzing the incidence of revision ABG in patients with and without postoperative CT scans for graft success evaluation.Setting:Academic tertiary care pediatric hospital.Patients:Eighty-seven patients with CLP or cleft lip and alveolus treated with autologous iliac crest bone grafting for alveolar clefts over a 10-year period (January 2009 to March 2019) with minimum 6-month follow-up. Fifty patients had postoperative CT evaluation; 37 did not.Interventions:Postoperative CT to determine ABG success, versus standard clinical examination and 2-dimensional radiographs.Main Outcome Measures:Requirement for revision ABG, defined as failure of the original graft by clinical or radiographic examination.Results:Fifty-eight percent of patients underwent a postoperative CT scan at median interval of 10 months after surgery. Patients with postoperative CT evaluation had a 44% rate of revision ABG (22/50) for inadequate graft take, compared to 5% (2/37) in patients without postoperative CT (P < .001; 95% CT, 31%-58% in the CT group, 1%-16% in the non-CT group).Conclusions:Computed tomography evaluation after ABG is associated with a significantly increased revision rate for inadequate graft take. The presence of a secondary palatal fistula at the time of original ABG is not associated with revision requirement. Lack of standardized dental and orthodontic records complicates the study of ABG outcomes and presents an area for systems-based improvement.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-01T08:59:02Z
      DOI: 10.1177/10556656211002689
       
  • The Throat Pack Debate: A Review of Current Practice in UK and Ireland
           Cleft Centers

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      Authors: Terry-Ann Curran, Nitisha Narayan, Lynn Fenner, Guy Thornburn, Marc C. Swan, Nefer Fallico
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:The use of throat packs during oropharyngeal surgery has long been a topic of debate among cleft surgeons. The advantage of inserting an absorbent tulle within the pharynx must be weighed against the risk of unintended retention postoperatively. Despite safety check mechanisms in place, retention may occur with potentially life-threatening consequences. We present a comprehensive review of throat pack use in all cleft units within the United Kingdom and Ireland.Methods:All 20 cleft surgery units in the United Kingdom and Ireland were surveyed on their use of throat packs in children aged 6 months to 2 years undergoing elective cleft palate surgery.Results:The response rate to the survey was 100%. Seventy-five percent of units currently use throat packs; in 40%, they are used in addition to cuffed endotracheal tubes (ETTs). Inclusion of the throat pack in the surgical swab count was perceived as the safest mechanism employed to avoid retention. 26.1% of respondents were aware of at least 1 incident of pack retention in their unit.Discussion/Conclusion:The reported UK and Irish experience demonstrates that three-quarters of units routinely use packs. Notably, a quarter of respondents to the survey have experience of an incident of throat pack retention. Nevertheless, the majority of respondents considered the perceived risk of retaining a pack to be low. The growing use of microcuffed ETTs in UK cleft units paired with a low incidence of perioperative complications when a throat pack is not introduced might prompt cleft surgeons to review routine pharyngeal packing.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-04-01T08:50:20Z
      DOI: 10.1177/10556656211000553
       
  • Evaluation of Velopharyngeal Closure Function With 4-Dimensional Computed
           Tomography and Assessment of Radiation Exposure in Pediatric Patients: A
           Cross-Sectional Study

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      Authors: Yoshikazu Kobayashi, Masanao Kobayashi, Daisuke Kanamori, Naoko Fujii, Yumi Kataoka, Koji Satoh, Yoshimi Sano, Satoshi Yoshioka, Ichiro Tateya, Hiroshi Toyama, Koichiro Matsuo
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Some patients with cleft palate (CP) need secondary surgery to improve functionality. Although 4-dimensional assessment of velopharyngeal closure function (VPF) in patients with CP using computed tomography (CT) has been existed, the knowledge about quantitative evaluation and radiation exposure dose is limited. We performed a qualitative and quantitative assessment of VPF using CT and estimated the exposure doses.Design:Cross-sectional.Setting:Computed tomography images from 5 preoperative patients with submucous CP (SMCP) and 10 postoperative patients with a history of CP (8 boys and 7 girls, aged 4-7 years) were evaluated.Patients:Five patients had undergone primary surgery for SMCP; 10 received secondary surgery for hypernasality.Main Outcome Measures:The presence of velopharyngeal insufficiency (VPI), patterns of velopharyngeal closure (VPC), and cross-sectional area (CSA) of VPI was evaluated via CT findings. Organ-absorbed radiation doses were estimated in 5 of 15 patients. The differences between cleft type and VPI, VPC patterns, and CSA of VPI were evaluated.Results:All patients had VPI. The VPC patterns (SMCP/CP) were evaluated as coronal (1/4), sagittal (0/1), circular (1/2), and circular with Passavant’s ridge (2/2); 2 patients (1/1) were unevaluable because of poor VPF. The CSA of VPI was statistically larger in the SMCP group (P = .0027). The organ-absorbed radiation doses were relatively lower than those previously reported.Conclusions:Four-dimensional CT can provide the detailed findings of VPF that are not possible with conventional CT, and the exposure dose was considered medically acceptable.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-31T09:00:15Z
      DOI: 10.1177/10556656211001732
       
  • Addition to the Toolbox of Surgical Techniques for Palatal Fistula Repair

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      Authors: Catherine de Blacam, David Orr
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      In response to the article by Rothermel and colleagues, the authors suggest the use of cancellous bone graft for repair of fistulae of the hard palate as an addition to the proposed toolbox.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-25T08:59:33Z
      DOI: 10.1177/10556656211003796
       
  • Intraoral Digital Impression for Speech Aid/Obturator in Children: Report
           of 2 Cases

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      Authors: Amara Abreu, Maria Helena Lima, Eric Hatten, Laura Klein, Daniel Levy-Bercowski
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Cleft patients may develop an abnormal opening (fistula) between the oral and the nasal cavities. Surgical repair minimizes the adverse effect on speech and feeding. However, an obturator prosthesis is a nonsurgical approach to help close the communication. The purpose of the case report presented is to show the clinical use of an intraoral digital impression in the fabrication of obturator/speech aid appliances in children with cleft lip and palate deformity. Minimal adjustments were needed, and patients and caregivers responded positively. Prostheses demonstrated good stability and retention at delivery. The use of digital technology seems to have several benefits as an alternative method for capturing impressions, especially in young children with cleft lip and palate deformity.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-25T08:59:12Z
      DOI: 10.1177/10556656211002686
       
  • Aesthetic Enhancement of the Vermilion Using Dermofat Graft in Patients
           With Cleft Lip Deformity

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      Authors: Kyung Hoon Chung, Karel A. deLeeuw, Hsiu-Hsia Lin, Lun-Jou Lo
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:There is an increasing patient expectation for better esthetics, manifesting through anatomical harmony, projection, and volume of the upper lip relative to the lower lip, in patients with cleft deformity. The aim of this study is to investigate the outcome of vermilion augmentation using autologous dermofat graft (DFG) to enhance the lip using both quantitative and qualitative assessment.Methods:Patients with secondary cleft lip deformity who received the surgical treatment in our institution from 2015 to 2018 were recruited. Panel assessment was performed on standardized preoperative and postoperative digital photographs. A patient questionnaire was used for the reported outcome. Image processing and analyses were applied to measure the lip change. Statistical analyses were performed.Results:A total of 91 patients were included. The mean age at operation was 22.7 years, and postoperative follow-up was 3.6 years. There were no complications in the study group. The panel assessment showed significant improvement (P < .00001) on upper lip vermilion in both frontal and lateral profile views. Ninety-five percent of patients reported improvement of the upper lip projection and volume. Quantitative image analysis showed an increase in the upper lip vermilion ratio in 97% of the cases in frontal views and improvement of the vermilion projection in 87% of the cases in the superimposed lateral views.Conclusions:The use of DFG is an effective and reproducible method for vermilion augmentation and aesthetic enhancement in patients presenting with upper lip insufficiency relative to the lower lip.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-25T08:58:14Z
      DOI: 10.1177/10556656211001727
       
  • Occlusal Outcome of Orthodontic Treatment for Patients With Complete Cleft
           Lip and Palate

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      Authors: Daniel Stonehouse-Smith, Aida N.A. Abd Rahman, Jeanette Mooney, Haydn Bellardie
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Aim:To assess occlusal outcomes of orthodontic treatment for patients with complete cleft lip and palate.Design:Retrospective assessment using the Peer Assessment Rating (PAR) index.Setting:Consecutive patients treated by one consultant orthodontist at a tertiary care cleft center.Participants:One hundred twenty-seven patients with either complete unilateral cleft lip and palate (UCLP) or bilateral cleft lip and palate (BCLP) consecutively treated with fixed appliances.Intervention:Fixed orthodontic appliance treatment and orthognathic surgery when required.Outcomes:The PAR index assessment was carried out by a calibrated-independent assessor. Treatment duration, the number of patient visits, and data on dental anomalies were drawn from patient records and radiographs.Results:One hundred two patients’ study models were assessed after exclusions. Mean start PAR score for UCLP (n = 71) was 43.9 (95% CI, 41.2-46.6, SD 11.5), with a mean score reduction of 84.3% (95% CI, 81.9-86.7, SD 10.1). The UCLP mean treatment time was 23.7 months with 20.1 appointments. Mean start PAR score for BCLP (n = 31) was 43.4 (95% CI, 39.2-47.6, SD 11.4), with a mean score reduction of 80.9% (95% CI, 76.3-85.5, SD 12.5). The BCLP mean treatment time was 27.8 months with 20.5 appointments.Conclusion:These results compare well with other outcome reports, including those for patients without a cleft, and reflect the standard of care provided by an experienced cleft orthodontist. As with high-volume surgeons, orthodontic treatment for this high need group is favorable when provided by a high-volume orthodontist. These findings may be used for comparative audit with similar units providing cleft care.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-24T08:57:39Z
      DOI: 10.1177/1055665621996116
       
  • Evaluation of Alt-RAMEC With Facemask in Patients With Unilateral Cleft
           lip and Palate (UCLP) Using Cone Beam Computed Tomography (CBCT) and
           Finite Element Modeling—A Clinical Prospective Study

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      Authors: Shalvi Singh, Puneet Batra, Sreevatsan Raghavan, Karan Sharma, Amit Srivastava
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Evaluate the effects of alternate rapid maxillary expansion and constriction (Alt-RAMEC) protocol on patients with unilateral cleft lip and palate (UCLP) along with maxillary protraction using cone beam computed tomography (CBCT) records and a finite element modeling (FEM) model.Materials and Methods:Twenty-five patients (aged 9-13 years) with UCLP having a GOSLON score 3 were selected for this prospective single-arm cohort study after obtaining necessary informed consent. A 9-week Alt-RAMEC protocol using a custom-made fan-shaped expansion screw was initiated, which was followed by 6-month facemask wear. Pre (T1) and posttreatment (T2) records including CBCT were taken and were evaluated for treatment changes in all 3 dimensions using paired t test. Additionally, a FEM model (ANSYS 15.0) using the Alt-RAMEC protocol was also analyzed for the displacement and stress on various craniofacial structures.Results:Significant forward movement of the maxilla along with clockwise rotation of mandible along with statistically significant increase in lower molar height and upper incisor proclination (P < .05) was observed. CBCT evaluation showed a statistical significant decrease in buccal alveolar bone thickness and increase in molar inclination (P < .05). Finite element modeling analysis highlighted that the maximum displacement occurred at the anterior region with stresses radiating till the anterior nasal spine, zygomatic arch, and sphenoid bone.Conclusion:Alt- RAMEC protocol combined with facemask protraction can lead to significant maxillary advancement, with some transient decrease in buccal bone thickness noted thereby warranting long-term studies to further assess its effect on the dentofacial apparatus.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-23T09:09:15Z
      DOI: 10.1177/10556656211000968
       
  • Clinical and Descriptive Study of Orofacial Clefts in Colombia: 2069
           Patients From Operation Smile Foundation

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      Authors: Alejandro Silva Espinosa, Julio Cesar Martinez, Yubahhaline Molina, María Alejandra Bermúdez Gordillo, Daniel Ramos Hernández, Daniela Zárate Rivera, Blanka Pacheco Olmos, Nathaly Ramírez, Liliana Arias, Andres Zarate, Q. Diana Marcela Diaz, Andrew Collins, Álvaro Mauricio Herrera Cepeda, Ignacio Briceño Balcazar
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To describe the population of patients with cleft lip and/or palate (CL/P) in terms of cleft phenotypes, gender, age, ethnic group, family history, clinical presentation (syndromic vs nonsyndromic), some environmental and behavioral factors, and some clinical features.Design:Descriptive retrospective study.Setting:Patients attending the genetics counseling practice in Operation Smile Foundation, Bogotá, Colombia, for over 8 years.Participants:No screening was conducted. All patients requiring clinical genetics assessment in Operation Smile Foundation were included in the study.Results:Left cleft lip and palate (CLP) and nonsyndromic forms were the most frequent types of malformations in this population. Psychomotor retardation and heart disease were the most frequent comorbidities in these patients. A low proportion of mothers exposed to passive smoking during pregnancy was observed and low birth weight accounted for an important number of cases. Aarskog, velocardiofacial, and orofaciodigital syndromes were the most frequent syndromic forms of CLP in this population.Conclusions:In this study, the most frequent type of CL/P was the nonsyndromic complete left CLP. Aarskog, velocardiofacial, and orofaciodigital syndromes were the most frequent syndromic forms of CL/P in this population.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-19T08:54:36Z
      DOI: 10.1177/10556656211000551
       
  • The Effect of Perichondrium and Graft Modification on the Viability of
           Conchal Cartilage Graft: An Experimental Study in Rabbit

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      Authors: Nathania Pudya Hapsari, Kristaninta Bangun, Parintosa Atmodiwirjo, Bambang Ponco, Tri Isyani Tungga Dewi, Jessica Halim
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Cartilage grafts are widely used in reconstructing nasal deformity for structural and aesthetic purposes. Despite being immunologically privileged, cartilage grafts are susceptible to volume loss with high risk of resorption over time. Therefore, experts opt for cartilage handling modification to resolve this issue through graft dicing, wrapping, or perichondrium preservation. This study will evaluate the effect cartilage graft preparations on graft viability.Design:Single-randomized post-test-only study design.Setting:Animal Hospital at Bogor Agricultural Institute.Participants:Six New Zealand, male, Hycole rabbits.Intervention:Conchal cartilage grafts were retrieved from 6 experimental rabbits and distributed into 3 treatment groups: diced cartilage graft (DC; control), one-sided perichondrium-attached scored cartilage (OPSC), and tube-shaped perichondrium-wrapped diced cartilage (TPDC).Main Outcome Measures:Macroscopic (weight and contour) and microscopic (chondroblast proliferation, graft thickness, apoptotic cells) evaluation through histological measures were recorded on week 12. Statistical analysis was done to compare between groups.Results:Diced cartilage and OPSC groups showed significant weight changes on week 12 (P < .05) with OPSC presenting with the biggest difference. Diced cartilage and OPSC group showed moderate cell proliferation on week 12 while TPDC displayed most abundant apoptotic cells (5.8%; P < .05). Diced cartilage group had the highest cartilage thickness ratio (P < .05).Discussion:Bare DC technique promoted graft thickness while perichondrium-attached scored cartilage showed the most abundant chondroblast proliferation and the least apoptotic cells. Perichondrium contributes to enhanced new cartilage formation.Conclusion:Diced cartilage graft is suitable for masking irregularity and volume augmentation, while perichondrium-attached cartilage graft is better for structural support in nasal reconstruction.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-19T08:53:57Z
      DOI: 10.1177/1055665621998173
       
  • Skeletal and Dental Correction and Stability Following LeFort I
           Advancement in Patients With Cleft Lip and Palate With Mild, Moderate, and
           Severe Maxillary Hypoplasia

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      Authors: Buddhathida Wangsrimongkol, Roberto L. Flores, David A. Staffenberg, Eduardo D. Rodriguez, Pradip. R. Shetye
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:This study evaluates skeletal and dental outcomes of LeFort I advancement surgery in patients with cleft lip and palate (CLP) with varying degrees of maxillary skeletal hypoplasia.Design:Retrospective study.Method:Lateral cephalograms were digitized at preoperative (T1), immediately postoperative (T2), and 1-year follow-up (T3) and compared to untreated unaffected controls. Based on the severity of cleft maxillary hypoplasia, the sample was divided into 3 groups using Wits analysis: mild: ≤0 to ≥−5 mm; moderate: −10 mm; and severe: ≤−10 mm.Participants:Fifty-one patients with nonsyndromic CLP with hypoplastic maxilla who met inclusion criteria.Intervention:LeFort I advancement.Main Outcome Measure:Skeletal and dental stability post-LeFort I surgery at a 1-year follow-up.Results:At T2, LeFort I surgery produced an average correction of maxillary hypoplasia by 6.4 ± 0.6, 8.1 ± 0.4, and 10.7 ± 0.8 mm in the mild, moderate, and severe groups, respectively. There was a mean relapse of 1 to 1.5 mm observed in all groups. At T3, no statistically significant differences were observed between the surgical groups and controls at angle Sella, Nasion, A point (SNA), A point, Nasion, B point (ANB), and overjet outcome measures.Conclusions:LeFort I advancement produces a stable correction in mild, moderate, and severe skeletal maxillary hypoplasia. Overcorrection is recommended in all patients with CLP to compensate for the expected postsurgical skeletal relapse.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-16T05:05:11Z
      DOI: 10.1177/1055665621996108
       
  • Day Care Surgery for Unilateral Alveolar Bone Grafting of Residual Clefts
           With Mandibular Symphyseal Bone—A Prospective Questionnaire Study

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      Authors: Martin Saaby, Sven Erik Nørholt, Annelise Küseler, Jens Hartlev
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate patients’ experiences of shortened hospitalization in relation to bone grafting of unilateral alveolar clefts with mandibular symphyseal bone grafts.Design:Prospective cohort questionnaire study.Setting:Department of Oral and Maxillofacial Surgery, Aarhus University Hospital, Denmark.Patients:Thirty-nine (male n = 26, female n = 13) patients with unilateral residual clefts after primary reconstruction, mean age 10.4 years.Interventions:All patients underwent a standardized procedure with closure of the alveolar defect using mandibular symphyseal bone grafts. Patients were discharged within a day after surgery. Within 24 hours after discharge from surgery, patients and their parents filled in a 14-item psychometric questionnaire assessing the quality of their recovery. For all questions, a visual analogue scale (VAS) (0-10) was used.Main Outcome Measure:Patients’ experience of shortened hospitalization in relation to secondary bone grafting of alveolar defects. Factors influencing the overall experience were measured using a VAS scale.Results:A significant correlation between “Information from the hospital” and feeling worried and anxious was found (P = .04). Additionally, the ability to sleep correlated with pain (P = .003) and with nausea (P = .001).Conclusions:Although this study included a limited number of patients, the findings suggest that treatment of alveolar residual cleft with bone grafting from the mandibular symphyseal region in a day care surgery setting is characterized by very high patient satisfaction.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-15T09:02:15Z
      DOI: 10.1177/1055665621999489
       
  • Cleft Care in a Developing Country: An Assessment of Knowledge and
           Attitudes of Patients/Parents of Children With an Orofacial Cleft to
           Orthodontic Treatment

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      Authors: Oluranti Olatokunbo daCosta, Ikenna Gerald Isiekwe, Chetachi Mariagoretti Ogbonna
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To assess the knowledge, awareness and attitude to orthodontic treatment of patients and parents of children with orofacial clefts, receiving comprehensive cleft care treatment.Design:Cross-sectional survey.Setting:A major tertiary health care facility in Nigeria, the Lagos University Teaching Hospital, Idi-araba, Lagos.Participants:Patients and parents of children with orofacial clefts receiving comprehnsive cleft care.Intervention:Patients and parents of children with orofacial clefts were asked to complete the survey. Interviewer-administered questionnaires were used.Results:A total of 123 patients participated in the study, 55.3% (68) were females and 44.7% (55) were males. The majority (85.3%, 114) of patients were children aged between 0 and 5 years, while only 7.3% (9) were adults, aged 18 years and above. About one-third (41) of the respondents indicated that they felt “their teeth was not straight.” Although 43.9% (54) reported that they knew that braces could be used to straighten teeth, only 13.8% of the respondents knew who an orthodontist was. Eighty percent (99) indicated that they would be willing to undergo treatment to straighten their teeth or their children/wards’ teeth.Conclusion:The majority of parents of children with clefts and adult patients attending the institution’s cleft clinic had limited knowledge of orthodontics and the orthodontic care required for patients with clefts, although they had a positive attitude toward orthodontic treatment. These findings will guide the center in planning patient-centered orthodontic care for patients with clefts while also highlighting areas of importance for patient counseling and education in the orthodontic care for these patients.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-10T09:22:33Z
      DOI: 10.1177/1055665621998179
       
  • Opioid Prescribing Practices in Cleft Lip and Cleft Palate Reconstruction

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      Authors: Reuben A. Falola, Jordan T. Blough, Jasson T. Abraham, Sebastian M. Brooke
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:Currently, there is no consensus regarding the role of opioids in the management of perioperative pain in children undergoing cleft lip/palate repair.Method:The present study evaluated opioid prescribing patterns of surgeon members within the American Cleft Palate-Craniofacial Association surgeons utilizing an anonymous survey.Results:Respondents performing cleft lip repair typically operate on patients 3 to 6 months of age (86%), admit patients postoperatively (82%), and discharge them on the first postoperative day (72%). Comparatively, respondents performed palatoplasty between the ages of 10 and 12 months (62%), almost always admit the patients (99%), and typically discharge on the first postoperative day (78%). Narcotics were more frequently prescribed after palatoplasty than after cleft lip repair, both for inpatients (66%; 49%) and at discharge (38%; 22%). Oxycodone was the most prescribed narcotic (39.1%; 41.4%), typically for a duration of 1 to 3 days (81.5%; 81.2%). All surgeons who reported changing their narcotic regimen (34.4% dose, 32.8% duration) after cleft lip repair, decreased both parameters from earlier to later in their career. Similarly, surgeons who changed the dose (32.2%) and duration (42.5%) of narcotics after palatoplasty, mostly decreased both parameters (96%). Additionally, physicians with >15 years of practice were less likely to prescribe opioids in comparison with colleagues with ≤15 years of experience. Ninety-two percent of respondents endorsed prescribing nonopioid analgesics after prescribing cleft surgery, most commonly acetaminophen (85.7%; 85.4%).Conclusion:Cleft surgeons typically prescribe opioids to inpatients and rarely upon discharge. Changes to opioid-prescribing patterns typically involved a decreased dose and duration.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-10T08:56:25Z
      DOI: 10.1177/1055665621990163
       
  • Three-Dimensional Assessment of Craniofacial Features in Patients With
           Down Syndrome During the Mixed Dentition Period: A Case-Control Study

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      Authors: Hideomi Takizawa, Masahiro Takahashi, Koutaro Maki
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:Down syndrome (DS) is a common congenital chromosomal disorder related to trisomy 21. Lateral cephalometric radiography studies have shown that patients with DS have characteristic craniofacial morphology; however, no 3-dimensional analysis studies have been performed to investigate the craniofacial features, including volumetric aspects, of patients with DS. The present study was performed to evaluate the craniofacial features, including volumetric aspects, of patients with DS and to compare these findings with control participants using cone beam computed tomography (CBCT).Materials and Methods:The study sample consisted of 12 patients with DS and 12 control participants. All participants were examined by means of CBCT; the resulting images were used for evaluation of maxillary and mandibular volume, cranial base, and craniofacial measurements. Differences between patients with DS and control participants were statistically analyzed using Student t test.Results:Compared to control participants, patients with DS exhibited statistically significant reductions in maxillary and mandibular volumes. Both sagittal and axial cranial base linear measurements were shorter in patients with DS than in control participants. In contrast, the cranial base angle was enhanced in patients with DS, compared with control participants. Moreover, condylion (Co)–gnathion, anterior nasal spine–menton, and Co-subspinale (point A) measurements were shorter in patients with DS than in control participants; the sella–nasion–mandibular plane angle was significantly reduced in patients with DS, compared with control participants.Conclusion:Our results suggest that patients with DS have distinct skeletal volume and craniofacial morphology features, relative to individuals without DS.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-09T09:45:14Z
      DOI: 10.1177/1055665621998181
       
  • Systematic Review of the Long-Term Effects of Presurgical Orthopedic
           Devices on Patient Outcomes

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      Authors: Katie Garland, Brendan McNeely, Luc Dubois, Damir Matic
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To perform a systematic review of the literature to identify the long-term effects of presurgical orthopedic (PSO) device use on patient outcomes.Design:A comprehensive literature review of Embase and Ovid databases was performed to identify all English-language publications related to unilateral cleft lip and palate, presurgical devices, and patient outcomes. Studies were excluded if they did not report patient outcomes beyond 2 years of age, did not describe the use of a PSO device, were case reports (n < 10), or were purely descriptive studies.Main Outcome Measures:Reported patient outcomes following the use of PSO devices.Results:Following a review of all articles by 2 independent reviews, 30 articles were selected for inclusion. Overall, there was no reported consensus as to the long-term effects of PSO devices. Furthermore, this study identified that only 10% of published research controlled for confounding factors that could influence the reported results. Confounding factors that were identified included different operating surgeon, different surgical protocols, and different rates of revision surgeries.Conclusions:Overall, this systematic review identified 2 important conclusions. Firstly, there is no consensus in the literature about the long-term effects of PSO devices on long-term patient outcomes. Secondly, research in this domain is limited by confounding factors that influence the applicability of the reported results.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-08T09:11:39Z
      DOI: 10.1177/1055665621998176
       
  • Treatment of Unilateral Cleft Lip and Palate Patient With Intraoral Tooth
           Tissue Borne Distractor and Facemask Therapy: A Case Report

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      Authors: Akansha Bansal Agrawal, Harshavardhan Kidiyoor, Anand K. Patil Morth
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      This case report demonstrates the successful use of intraoral distractor/hygenic rapid expander (HYRAX) for rapid maxillary expansion in anteroposterior direction with an adjunctive use of face mask therapy for anterior orthopedic traction of maxillary complex in a cleft patient with concave profile. The patient was a 13-year-old girl who reported with a chief complaint of backwardly positioned upper jaw and a severely forward positioned lower jaw. Therefore, a treatment was chosen in which acrylic bonded rapid maxillary expansion was done with tooth tissue borne intraoral distractor/HYRAX having a different activation schedule along with Dr Henri Petit facemask to treat maxillary retrognathism. As a result, crossbite got corrected and attained a positive jet with no bone loss in cleft area over a period of 5 months which was followed by fixed mechanotherapy achieving a well settled occlusion in 1 year. After completion of expansion and fixed mechanotherapy, ANB became +1 post-treatment which was −4 pretreatment. The prognathic profile was markedly improved by expansion and taking advantage of the remaining growth potential, thus minimizing the chances of surgery later in life. This provided a viable alternative to orthognathic surgery with good long-term stability.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-08T09:00:19Z
      DOI: 10.1177/1055665621993366
       
  • Impact of Surgery for Velopharyngeal Insufficiency on Eustachian Tube
           Function in Children: Pharyngeal Flap Versus Sphincter Pharyngoplasty

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      Authors: Colin Fuller, Kesley Brown, Olivia Speed, James Gardner, Ashlen Thomason, John Dornhoffer, Adam B Johnson, Larry Hartzell
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Velopharyngeal insufficiency (VPI) is a common speech disorder in patients with a history of cleft palate (CP) or 22q11.2 deletion syndrome. Pharyngeal flap (PF) and sphincter pharyngoplasty (SP) are 2 common surgeries to treat this disorder by decreasing unwanted nasal air emission and hypernasal resonance. Because Eustachian tube dysfunction (ETD) in patients with CP may be more frequent after surgery for VPI, we examined whether ETD was associated with either type of surgery.Design:Retrospective cohort study.Setting:Children’s hospital-based tertiary referral center.Patients:A total of 225 children with VPI who underwent primary PF (201) or SP (24) between 2006 and 2017.Outcome measures:We examined differences in risk of ETD according to both surgical groups and proxies for postoperative nasal obstruction. These proxies included postoperative resonance measures and development of obstructive sleep apnea (OSA).Results:Both surgical groups had similar preoperative measures, except the PF group had higher hypernasality by PSA. Postoperatively, the PF group demonstrated lower hypernasal resonance by nasometry and PSA. There were no differences between PF and SP groups with regard to ETD. Proxies for postoperative nasal obstruction also were not predictive of postoperative ETD. Degree of CP and younger age were found to be risk factors for ETD.Conclusion:There was no significant difference in the effects of PF and SP on ETD in this study. Neither lower hypernasality nor incidence of OSA had any impact on ETD. Degree of CP and younger age were the only significant risk factors for ETD that this study identified.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-08T08:59:19Z
      DOI: 10.1177/1055665621991740
       
  • Clinical Correction of Complete Median Cleft of the Mandible and Lower
           Lip: A 17-Year Follow-Up of a Case Report With Literature Review

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      Authors: Weidong Liu, Li Ma, Shizhou Zhang, Tengda Zhao
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      The median cleft of the mandible and lower lip is an extremely rare congenital maxillofacial deformity, and the therapeutic options are controversial. To evaluate the clinical characteristics and identify a better choice of treatment modes used among us and others, we reviewed 34 relevant literature and herein describe a 17-year follow-up of a case with a median cleft of the mandible and lower lip. Based on the literature and our case with good functional and aesthetical outcomes, we propose a prospective clinical treatment: Patients of Tessier 30 cleft associated with cleft of the mandible could undergo mandibular repair after puberty in conditions of a good occlusal relationship and normal maxillofacial development, even with mild masticatory dysfunction.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-08T08:58:20Z
      DOI: 10.1177/1055665621990170
       
  • Barriers and Facilitators to the International Implementation of
           Standardized Outcome Measures in Clinical Cleft Practice

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      Authors: Inge Apon, Carolyn R. Rogers-Vizena, Maarten J. Koudstaal, Alexander C. Allori, Petra Peterson, Sarah L. Versnel, Jessily P. Ramirez
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To identify barriers and facilitators to international implementation of a prospective system for standardized outcomes measurement in cleft care.Design:Cleft teams that have implemented the International Consortium for Health Outcomes Measurement Standard Set for cleft care were invited to participate in this 2-part qualitative study: (1) an exploratory survey among clinicians, health information technology professionals, and project coordinators, and (2) semistructured interviews of project leads. Thematic content analysis was performed, with organization of themes according to the dimensions of the reach, effectiveness, adoption, implementation and maintenance (RE-AIM) framework: reach, effectiveness, adoption, implementation, and maintenance.Results:Four cleft teams in Europe and North America participated in this study. Thirteen participants completed exploratory questionnaires and 5 interviewees participated in follow-up interviews. Survey responses and thematic content analysis revealed common facilitators and barriers to implementation at all sites. Teams reach patients either via email or during the clinic visit to capture patient-reported outcomes. Adopting routine data collection is enhanced by aligning priorities at the organizational and cleft team level. Streamlining workflows and developing an efficient data collection platform are necessary early on, followed by pilot testing or stepwise implementation. Regular meetings and financial resources are crucial for implementing, sustaining, analyzing collected data, and providing feedback to health care professionals and patients. Fostering patient-centered care was articulated as a positive outcome, whereas time presented challenges across all RE-AIM dimensions.Conclusions:Identified themes can inform ongoing implementation efforts. Intentionally investing time to lay a sound foundation early on will benefit every phase of implementation and help overcome barriers such as lack of support or motivation.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-05T08:55:44Z
      DOI: 10.1177/1055665621997668
       
  • Comparing the Treatment Outcomes of Absorbable Sutures, Nonabsorbable
           Sutures, and Tissue Adhesives in Cleft Lip Repair: A Systematic Review

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      Authors: Uchenna P. Egbunah, Olawale Adamson, Azeez Fashina, Adegbayi A. Adekunle, Olutayo James, Wasiu L. Adeyemo
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objectives:To examine the literature and synthesize the available reports for the best possible option between absorbable, nonabsorbable, and tissue adhesives in cleft lip skin closure.Design:We conducted systematic searches for randomized controlled trials and controlled clinical trials in PubMed, Cochrane, Ovid Medline, and OpenGrey databases. Identified studies were retrieved and assessed for eligibility. All statistical analyses were done with Revman, version 5.4.Interventions:The intervention considered in this systematic review were techniques of cleft lip repair using resorbable sutures, nonabsorbable sutures, medical adhesives, or any combination of these.Outcome Measures:The primary outcomes assessed in the trials had to include any combination of the following: wound healing cosmesis and wound healing complications. While secondary outcomes considered were quality of life, direct and indirect costs to patients and health services, and participant satisfaction.Results:Only 6 studies met all inclusion criteria and were selected for qualitative analysis. A more favorable wound healing cosmesis was seen when nonabsorbable suture was used in cleft lip repair compared to absorbable sutures and tissue adhesives (CI, 0.65-4.35). This advantage was overshadowed by the significantly higher prevalence of postoperative complications when nonabsorbable sutures are used.Conclusion:Although the results point to more favorable cosmesis with nonabsorbable sutures and an overall more favorable outcome with either absorbable sutures or tissue adhesives, the 6 selected studies were assessed at an unclear risk of bias; therefore, the results of this study should be interpreted with caution and regarded as low-certainty evidence.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-03T10:03:27Z
      DOI: 10.1177/1055665621996107
       
  • A Cleft-Customized Occlusal Rating System to Assess Orthodontic Occlusal
           Improvement in Patients With Unilateral Cleft Lip and Palate

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      Authors: Fabio Henrique de Sa Leitao Pinheiro, Carolina Martins Frota, Daniela Gamba Garib, Renata Sathler, Terumi Okada Ozawa, Rita de Cassia Moura Carvalho Lauris, Renata Mayumi Kato, Érika Tiemi Kurimori
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:This study aimed to develop a new method to quantify occlusal improvement in patients with unilateral cleft lip and palate (UCLP) who had undergone orthodontic treatment and to evaluate its reproducibility.Design:A panel of orthodontists decided on the relevance of different occlusal features to score initial and final 3-dimensional study models and panoramic radiographs. A subsequent subjective analysis was later performed by a local orthodontic panel.Setting:The sample was obtained from the orthodontic clinical archives of a hospital known for the treatment of patients with craniofacial differences.Patients:Thirty-one nonsyndromic patients, 17 males and 14 females, were randomly selected according to preestablished inclusion/exclusion criteria.Interventions:The records corresponded to the period during which the patients were treated with conventional multibracket mechanics and adjunctive restorative procedures.Main Outcome/Measures:The intraclass correlation coefficient measured intraexaminer and interexaminer agreements. The Spearman correlation test assessed the relationship between the local orthodontic panel perception and the improvement scores.Results:Inter- and intra-rater ICCs varied between fair/good to excellent. There was a strong correlation between the Cleft-Customized Occlusal Rating system classification of occlusal improvement and the local orthodontic panel’s perception, thereby enabling the utilization of the interpretation scale by the panel.Conclusions:The method showed to be a useful tool in quantifying and classifying occlusal improvement in this specific population. As any other method, some limitations apply and need to be accounted for.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-03T09:28:53Z
      DOI: 10.1177/1055665621995313
       
  • Percutaneous Enteral Feeding in Patients With 22q11.2 Deletion Syndrome

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      Authors: Bridget Ebert, Noelle Morrell, Hanan Zavala, Sivakumar Chinnadurai, Robert Tibesar, Brianne Barnett Roby
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To describe the incidence of percutaneous enteral feeding in patients with 22q11.2 deletion syndrome (22q11.2 DS) and determine factors associated with the need for percutaneous enteral feeding tube placement.Design:Retrospective chart review.Methods:The records of a 22q11.2 DS clinic and pediatric otolaryngology clinic at a tertiary pediatric hospital were reviewed from January 1, 2009, to December 31, 2019. All patients with confirmed 22q11.2 deletion were identified. Cardiac, otolaryngological, and feeding characteristics were recorded along with surgical history. A patient was defined to have a G-tube if the history was significant for any percutaneous gastric feeding tube placement, including a gastrostomy tube, gastrostomyjejunostomy tube, or a Mickey button.Results:One hundred ninety patients with confirmed 22q11.2 DS by genetic testing were included. Thirty-three percent (n = 63) required G-tube placement. G-tube placement was associated with cardiac diagnosis (P < .01), history of cardiac surgery (P < .01), aspiration (P < .01), nasopharyngeal reflux (P < .01), subglottic stenosis (P < .01), laryngeal web (P = .003), and tracheostomy (P < .01). This suggests these conditions are associated with higher rates of G-tube placement in the 22q11.2 DS population.Conclusions:Patients with 22q11.2 DS often require supplemental nutritional support in the form of G-tube feeding, most often in the first year of life. Congenital heart abnormalities and surgery along with tracheostomy, subglottic stenosis, laryngeal web, aspiration, and nasopharyngeal reflux are significantly associated with the need for G-tube placement. Understanding associations between comorbid conditions and G-tube placement, especially those involving the head and neck, may assist with counseling of patients with 22q11.2 DS.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-02T09:36:03Z
      DOI: 10.1177/1055665621996117
       
  • Factors Associated With Delay in Cleft Surgery at a Tertiary Children’s
           Hospital in a Major US Metropolitan City

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      Authors: Jacqueline Stoneburner, Naikhoba C. O. Munabi, Eric S. Nagengast, Madeleine S. Williams, Pedram Goel, Allyn Auslander, Lori K. Howell, Jeffrey A. Hammoudeh, Mark M. Urata, William P. Magee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To identify factors associated with late cleft repair at a US tertiary children’s hospital.Design:Retrospective study of children with CL/P using Children’s Hospital Los Angeles (CHLA) records.Setting:US tertiary children’s hospital.Patients/Participants:Patients undergoing primary CL or CP repair at CHLA from 2009 to 2018.Main Outcome Measures:Proportion of children who had delayed primary CL repair or CP repair using CHLA and American Cleft Palate-Craniofacial Association (ACPA) guidelines and factors associated with late surgery.Results:In total, 805 patients—503 (62.5%) who had CL repair, 302 (37.5%) CP repair—were included. Using CHLA protocol, 14.3% of patients seeking CL repair had delayed surgery. Delay was significantly associated with female gender, non-Hispanic ethnicity, Spanish primary language, government insurance, bilateral cleft, cleft lip and palate (CLP), and syndromic diagnosis. Using ACPA guidelines, 5.4% had delayed surgery. Female gender and syndromic diagnosis were significantly associated with delay and remained significant after adjustment for confounders in multivariate models. For CP repair, 60.3% of patients had delayed surgery using CHLA protocol. Cleft lip and palate diagnosis, complete cleft, syndromic diagnosis, and longer travel distance were significantly associated with delay. Using ACPA guidelines, 28.5% had delayed surgery; however, significant association with patient variables was not consistently observed.Conclusions:Delay in cleft surgery occurs most often for patients seeking CP repair and is associated with female gender, non-Hispanic ethnicity, Spanish language, government insurance, and bilateral CL, CLP, or syndromic diagnoses. Initiatives should aim to optimize cleft surgery delivery for these subpopulations.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-03-02T09:34:35Z
      DOI: 10.1177/1055665621989508
       
  • Long-Term Treatment Outcomes of Primary Alveolar Bone Grafts for Alveolar
           Clefts: A Qualitative Systematic Review

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      Authors: M. Kristine Carbullido, Riley A. Dean, George N. Kamel, Greta L. Davis, Michael Hornacek, Rachel M. Segal, Emily Ewing, Samuel H. Lance, Amanda A. Gosman
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:Alveolar bone grafting is utilized to manage alveolar clefts in patients with cleft lip and palate. However, the timing of bone grafting is variable with conflicting evidence supporting the use of primary alveolar bone grafting (PABG) in clinical practice.Primary Aim:To provide a qualitative systematic review analysis of long-term outcomes after PABG.Materials and Methods:A qualitative systematic review was performed following the Cochrane Handbook and reported using Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Summative findings were evaluated using Confidence in the Evidence from Reviews of Qualitative research to assess the quality of evidence supporting the findings.Results:After removing duplication, 2182 publications were identified, and 2131 were excluded after screening through titles and abstracts. Inclusion criteria for this study included patients who underwent PABG at 24 months of age or younger and a minimum of 5 year follow-up. Thirty-two publications met the inclusion criteria and were included for qualitative analysis. Primary outcome measures included cephalometric analysis, bone graft survival, occlusal analysis, hypomineralization, tooth eruption, radiograph analysis, and arch relationships. Four assessment themes were characterized from the systematic review: (1) bone graft survival, (2) craniofacial skeletal relationships, (3) occlusion and arch forms, and (4) recommendations for utilizing PABG in practice.Conclusion:The reported systematic review provides evidence that performing PABG leads to poor long-term outcomes related to bone graft survival and maxillary growth restriction despite some reported positive outcomes.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-26T07:32:35Z
      DOI: 10.1177/1055665621995047
       
  • Changes in the Oral Health-Related Quality of Life in Infants With Cleft
           Lip and/or Palate Before and After Surgical Treatment

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      Authors: Roxana Patricia López Ramos, Daniel José Blanco Victorio, Gilmer Torres Ramos, Mónica J. Pajuelo, Jenny Abanto
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To assess oral health-related quality of life (OHRQoL) changes before and after the primary surgical treatment in infants with cleft lip and/or palate (CL/P).Design:Quasi-experimental study.Methods:A total of 106 infants with CL/P younger than 2 years undergoing primary surgical treatment in the Plastic Surgery Service of the Instituto Nacional de Salud del Niño in Peru. The parent/caregiver answered a questionnaire about OHRQoL named the Peruvian version of the Early Childhood Oral Health Impact Scale (P-ECOHIS) in the pretreatment (baseline) and follow-up post-treatment. The total score of P-ECOHIS and their 2 sections (child impact and family impact) in the baseline and each follow-up period post-treatment scores were assessed. As well as, the type of the CL/P on OHRQoL, standardized effect sizes (ES) based on mean total change scores (difference between baseline and 12th month) were analyzed.Results:Improvements in infant’s OHRQoL after treatment were reflected in each follow-up period P-ECOHIS score compared to the baseline score. The total P-ECOHIS scores decreased significantly from 28.07 (baseline) to 7.7 (12th month; P < .0001), as did the individual domain scores (P < .0001). There were significant differences in the baseline and follow-up post-treatment scores of infants who reported improvement of the OHRQoL (P < .0001). The ES was large (3.79). The cleft lip had an improvement in the OHRQoL at 12th month post-treatment (P < .0001).Conclusions:Primary surgical post-treatment resulted in significant improvement of the infant’s OHRQoL with CL/P.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-25T09:16:37Z
      DOI: 10.1177/1055665621993282
       
  • Response to Pulp Vitality Tests in Teeth Submitted to Orthodontic
           Movement, Adjacent to the Cleft Area

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      Authors: Claudia Ramos Pinheiro, Lidiane de Castro Pinto, José Francisco Mateo-Castillo, Viviane da Silva Siqueira, Daniela Garib
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To evaluate the pulp vitality in teeth adjacent to the cleft area submitted to orthodontic movement into the alveolar graft area in individuals with complete unilateral cleft lip and palate (CUCLP).Design:Cold sensitivity, vertical, and horizontal percussion tests were conducted on the teeth adjacent to the cleft and the contralateral teeth.Setting:Endodontics Sector in the Hospital for Rehabilitation of Craniofacial Anomalies, University of São Paulo (HRAC/USP).Patients:One hundred patients with CUCLP and hypodontia of the upper lateral incisor in orthodontic movement and after successful alveolar bone graft in the cleft area.Main Outcome Measures:The cleft study group (SG) was composed of 200 teeth, adjacent to the cleft area. The control group (CG) consisted of 200 contralateral teeth. Statistical analysis was performed using the chi-square test for comparisons between groups (P < .05).Results:In the SG, 82.0% of teeth presented positive response to the cold sensitivity testing, 13.5% had negative response, and 4.5% had marked response, with statistically significant difference in relation to the CG. The vertical and horizontal percussion tests on teeth in the SG revealed the same results, in which 95.0% presented negative response and 5.0% responded positively, without significant difference compared to teeth in the CG, for both tests.Conclusions:Teeth adjacent to the cleft area presented changes in the physiological conditions of the pulp, which were observed by reduction of positive response to the cold sensitivity testing or presence of pulp hypersensitivity in cases of marked response.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-24T09:35:43Z
      DOI: 10.1177/1055665621996115
       
  • Perceptions Regarding the Academic and Cognitive Performance of
           Individuals With Cleft Lip and/or Palate

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      Authors: İlkem Kara, Aydan Baştuğ Dumbak, Maviş Emel Kulak Kayıkcı
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Introduction:Factors such as teachers’ appropriate support and social interactions have an impact on the academic performance of children with cleft lip and/or palate (CL/P). This study was designed to investigate the perceptions of the teachers and the general public about the academic and cognitive performance of individuals with CL/P.Methods:This study was included 360 (male/female = 102/258) teachers and 640 (male/female = 259/381) participants that represent the general public. Anonymized web-based and paper-and-pencil self-administered questionnaire that included multiple-choice and yes/no questions were administered. Within-group differences and intergroup differences were analyzed in terms of academic and cognitive performance.Results:Most of the teachers and the general public indicated that the academic and cognitive performance of individuals with CL/P is the same as their unaffected peers. A significantly higher proportion of the teachers indicated that the academic performance of children with CL/P is the same as their unaffected peers than the general public.Conclusion:Considering that the general public’s attitudes and appropriate teacher support are crucial to prevent adverse impacts on the lives of individuals with CL/P, it is important to support teachers with the appropriate information and to encourage the public to recognize that everybody with a facial difference should be treated as an individual rather than a disability.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-24T09:30:03Z
      DOI: 10.1177/1055665621995308
       
  • Clefts and Dental General Anesthesia: The South Thames Experience

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      Authors: Maalini Patel, Mina Vaidyanathan, Nabina Bhujel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background:Children with clefts have high dental needs and often require a dental general anesthesia (DGA) for the management of their dentition.Objective:To assess the number of children with clefts requiring a pediatric dental consultant led DGA in the South Thames Cleft Service over a 3-year period.Design:Data were collected retrospectively over a 36-month period.Results:Sixty-five children with cleft lip and/or palate required a DGA with the South Thames Cleft Service. The average age of the child was 7 years, and the average number of teeth treated under the general anesthetic was 9. Families traveled an average of 21 miles to access their child’s care, and the referral-to-treat time was on average 14 weeks.Conclusion:Results highlight the need for DGA provision for children with clefts. Cleft centers need sufficient capacity for this service to prevent long wait times, and in view of distances traveled shared, care should be implemented with local providers.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-23T09:51:03Z
      DOI: 10.1177/1055665621995044
       
  • A Quantitative Analysis of Facial Asymmetry in Torticollis Using
           3-Dimensional Photogrammetry

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      Authors: Vanessa M. Baratta, Olivia E. Linden, Margaret E. Byrne, Stephen R. Sullivan, Helena O. Taylor
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To assess whether children with torticollis have quantifiably greater facial asymmetry than their age-matched controls using 3-dimensional (3D) photogrammetry.Design:We retrospectively analyzed patients diagnosed with torticollis and age-matched volunteers who underwent 3D photogrammetry of their faces. We calculated the root mean square deviation (RMSD) between native and reflected facial images, as a measure of asymmetry. Two observers independently measured RMSD values for all study participants. The Spearman correlation coefficient evaluated interobserver reliability. The Wilcoxon rank-sum test with Bonferroni adjusted P values for multiple comparisons.Setting:Institutional.Participants:Twenty patients diagnosed with torticollis and 12 age-matched volunteers. Patients were analyzed on a computer database and volunteers were selected and consented in the hospital. We excluded patients with a history of facial trauma, facial operations, or other craniofacial diagnoses.Interventions:Facial surface scans were obtained using the Canfield Vectra stereophotogrammetry system. The technology captures surface anatomy without radiation.Main Outcome Measures:RMSD comparisons between patients with torticollis and age-matched controls.Results:Compared to controls, patients with torticollis had statistically significant greater full face, upper third, and middle third facial asymmetry. There was a trend toward greater asymmetry of the lower facial third.Conclusions:We used 3D photogrammetry to quantitate facial asymmetry from torticollis. We found greater asymmetry in patients with torticollis than in their unaffected peers. All areas of the face appeared to be affected, though the asymmetry in the lower facial third just failed to reach significance.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-17T09:44:16Z
      DOI: 10.1177/1055665621993284
       
  • Parenting Children With a Cleft Lip With or Without Palate or a Visible
           Infantile Hemangioma: A Cross-Sectional Study of Distress and Parenting
           Stress

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      Authors: M. van Dalen, W.H. Leemreis, V. Kraaij, P.C.J. De Laat, S.G.M.A. Pasmans, S.L. Versnel, M.J. Koudstaal, M.H.J. Hillegers, E.M.W.J. Utens, J.M.E. Okkerse
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Parents of children with a medical condition and a visible difference can experience challenging situations. We evaluated distress and parenting stress in parents of children with a cleft lip with or without cleft palate (CL±P) or a visible infantile hemangioma (IH).Setting:This cross-sectional study took place in an academic medical hospital in Rotterdam, the Netherlands.Participants:Three-hundred nine parents (mean age = 40.30, 56.00% mothers) of children with CL±P and 91 parents (mean age = 36.40, 58.24% mothers) of children with IH.Main Outcome Measures:The Dutch version of the Parenting Stress Index – Short Form and the subscales Anxiety, Depression, and Hostility of the Symptom Checklist – 90.Results:One sample t tests and mixed linear modeling were used. On average, parents of children with CL±P and of children with IH showed significantly lower parenting stress compared to normative data. Anxiety was significantly lower in parents of children with CL±P than that in the norm group. Visibility of the condition was not related to distress or parenting stress. Child behavioral problems were positively related to parenting stress, depression, and hostility.Conclusions:Parents of children with CL±P and IH report less distress and parenting stress compared to the norm. On average, these parents seem well adjusted. A practical implication is to monitor parents of children with behavioral problems.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-15T09:33:07Z
      DOI: 10.1177/1055665621993298
       
  • Administration of Single-Dose Antibiotic Does Not Decrease Oronasal
           Fistula Rates After Primary Palatoplasty

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      Authors: Ishwarya Shradha Mamidi, Esperanza Mantilla-Rivas, Brynne A. Ichiuji, Md Sohel Rana, Karen I. Ramirez, Jason R. Stein, Marudeen Aivaz, Monica Manrique, Jennifer McGrath, Gary F. Rogers, Albert K. Oh
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Oronasal fistula (ONF) is a known complication after primary palatoplasty (PP). Studies investigating the effect of perioperative antibiotics on fistula rates after PP are limited by inadequate sample size or reliance on self-reporting through national databases. In this study, the authors evaluated the association between single-dose perioperative antibiotics and postoperative fistula rates after PP at a single institution.Design:A retrospective study.Participants:Children younger than 2 years who underwent PP from April 2009 to September 2019 were included.Interventions:Patients were divided into 2 categories: Group 1 received a single intraoperative dose of IV antibiotic, while group 2 did not.Main Outcome Measure(s):Outcome measures included ONF formation, length of stay (LOS), and 30-day readmission rates. Multivariable firth logistic regression, quantile regression, and χ2 tests were performed.Results:Of the 424 patients, 215 and 209 patients were in groups 1 and 2, respectively. The overall ONF rate was 1.9% among all patients. Patients in group 1 experienced an ONF rate of 3.3%, while patients in group 2 had an ONF rate of 0.5%. After correcting for confounding variables, the difference in ONF rates was not statistically different (P = .68). Median LOS was 35.7 hours and 35.5 hours (P = .17), while the rate of readmission within 30 days was 4.7% and 2.4% for group 1 and 2, respectively (P = .96).Conclusions:Administration of a single-dose perioperative antibiotic did not decrease fistula formation after PP, nor did it affect the patient’s LOS or 30-day readmission rate.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-15T09:32:02Z
      DOI: 10.1177/1055665621992653
       
  • Palatoplasty for the Patient With Campomelic Dysplasia—Report of a Case
           and Review of the Literature

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      Authors: Kaya Narimatsu, Akihiko Iida, Takanori Kobayashi
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Campomelic dysplasia (CMPD) is a skeletal disorder resulting from SOX9 gene mutations. Palatoplasty is rare due to a high lethality rate in infants from respiratory distress. Our patient had characteristic symptoms of CMPD, including short bowed limbs, macrocephaly, low-set ears, short palpebral fissures, hypertelorism, a flat nasal bridge, a long philtrum, micrognathia, and a cleft palate. We performed a Furlow palatoplasty when the patient was 2 years 9 months of age, after respiratory conditions had stabilized. We reviewed the literature of CMPD cases that underwent palatoplasty and discussed the optimal timing and surgical methods.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2021-02-12T10:23:35Z
      DOI: 10.1177/1055665621992654
       
  • Masticatory Function Assessment of Adult Patients With Cleft Lip and
           Palate After Orthodontic Treatment

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