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ORTHOPEDICS AND TRAUMATOLOGY (150 journals)                     

Showing 1 - 152 of 152 Journals sorted alphabetically
Acta Orthopaedica     Open Access   (Followers: 32)
Advances in Orthopedics     Open Access   (Followers: 9)
American Journal of Orthodontics and Dentofacial Orthopedics     Hybrid Journal   (Followers: 9)
American Journal of Orthopedics     Partially Free   (Followers: 3)
Archives of Orthopaedic and Trauma Surgery     Hybrid Journal   (Followers: 9)
Archives of Osteoporosis     Hybrid Journal   (Followers: 1)
Arthritis und Rheuma     Hybrid Journal  
Arthroplasty Today     Open Access   (Followers: 1)
Australasian Musculoskeletal Medicine     Full-text available via subscription   (Followers: 5)
BMC Musculoskeletal Disorders     Open Access   (Followers: 29)
Bone & Joint 360     Full-text available via subscription   (Followers: 18)
Bone Research     Hybrid Journal   (Followers: 2)
Burns & Trauma     Open Access   (Followers: 11)
Cartilage     Hybrid Journal   (Followers: 5)
Case Reports in Orthopedic Research     Open Access  
Case Reports in Orthopedics     Open Access   (Followers: 6)
Chinese Journal of Traumatology     Open Access  
Cleft Palate-Craniofacial Journal     Hybrid Journal   (Followers: 8)
Clinical Medicine Insights : Arthritis and Musculoskeletal Disorders     Open Access   (Followers: 3)
Clinical Orthopaedics and Related Research     Hybrid Journal   (Followers: 78)
Clinical Trials in Orthopedic Disorders     Open Access   (Followers: 1)
Concussion     Open Access  
Craniomaxillofacial Trauma and Reconstruction     Hybrid Journal   (Followers: 1)
Current Orthopaedic Practice     Hybrid Journal   (Followers: 14)
Current Reviews in Musculoskeletal Medicine     Open Access   (Followers: 13)
Der Orthopäde     Hybrid Journal   (Followers: 6)
Die Wirbelsäule     Hybrid Journal  
Duke Orthopedic Journal     Open Access   (Followers: 5)
East African Orthopaedic Journal     Full-text available via subscription  
EFORT Open Reviews     Open Access   (Followers: 1)
Egyptian Orthopaedic Journal     Open Access   (Followers: 1)
EMC - Técnicas Quirúrgicas - Ortopedia y Traumatología     Full-text available via subscription  
EMC - Tecniche Chirurgiche - Chirurgia Ortopedica     Full-text available via subscription  
Ergonomics     Hybrid Journal   (Followers: 22)
European Journal of Orthopaedic Surgery & Traumatology     Hybrid Journal   (Followers: 9)
European Journal of Podiatry / Revista Europea de Podología     Open Access   (Followers: 1)
European Spine Journal     Hybrid Journal   (Followers: 24)
Foot & Ankle International     Hybrid Journal   (Followers: 10)
Foot & Ankle Orthopaedics     Open Access   (Followers: 3)
Gait & Posture     Hybrid Journal   (Followers: 17)
Geriatric Orthopaedic Surgery Rehabilitation     Open Access   (Followers: 5)
Global Spine Journal     Open Access   (Followers: 12)
Hip International     Hybrid Journal  
Indian Journal of Orthopaedics     Open Access   (Followers: 8)
Informationen aus Orthodontie & Kieferorthopädie     Hybrid Journal  
Injury     Hybrid Journal   (Followers: 20)
International Journal of Orthopaedic and Trauma Nursing     Hybrid Journal   (Followers: 11)
International Journal of Orthopaedic Surgery     Open Access   (Followers: 5)
International Journal of Orthopaedics     Open Access   (Followers: 2)
International Journal of Research in Orthopaedics     Open Access  
International Musculoskeletal Medicine     Hybrid Journal   (Followers: 7)
International Orthopaedics     Hybrid Journal   (Followers: 18)
JAAOS : Global Research & Reviews     Open Access   (Followers: 1)
JBJS Journal of Orthopaedics for Physician Assistants     Hybrid Journal  
JBJS Reviews     Full-text available via subscription   (Followers: 11)
JOR Spine     Open Access   (Followers: 3)
Journal de Traumatologie du Sport     Full-text available via subscription   (Followers: 2)
Journal für Mineralstoffwechsel & Muskuloskelettale Erkrankungen     Hybrid Journal  
Journal of Bone and Joint Diseases     Open Access   (Followers: 4)
Journal of Bone and Joint Infection     Open Access   (Followers: 1)
Journal of Brachial Plexus and Peripheral Nerve Injury     Open Access   (Followers: 4)
Journal of Cachexia, Sarcopenia and Muscle     Open Access   (Followers: 2)
Journal of Children's Orthopaedics     Open Access   (Followers: 10)
Journal of Clinical Orthopaedics and Trauma     Hybrid Journal   (Followers: 5)
Journal of Experimental Orthopaedics     Open Access   (Followers: 8)
Journal of Hand Surgery (European Volume)     Hybrid Journal   (Followers: 44)
Journal of Head Trauma Rehabilitation     Hybrid Journal   (Followers: 17)
Journal of Musculoskeletal Research     Hybrid Journal   (Followers: 9)
Journal of Orofacial Orthopedics / Fortschritte der Kieferorthopädie     Hybrid Journal  
Journal of Orthodontic Science     Open Access   (Followers: 2)
Journal of Orthopaedic & Sports Physical Therapy     Full-text available via subscription   (Followers: 70)
Journal of Orthopaedic Association of South Indian States     Open Access   (Followers: 5)
Journal of Orthopaedic Diseases and Traumatology     Open Access   (Followers: 5)
Journal of Orthopaedic Reports     Full-text available via subscription   (Followers: 12)
Journal of Orthopaedic Research     Hybrid Journal   (Followers: 29)
Journal of Orthopaedic Science     Hybrid Journal   (Followers: 4)
Journal of Orthopaedic Surgery     Open Access   (Followers: 1)
Journal of Orthopaedic Surgery and Research     Open Access   (Followers: 8)
Journal of Orthopaedic Translation     Open Access  
Journal of Orthopaedic Trauma     Hybrid Journal   (Followers: 15)
Journal of Orthopaedics     Full-text available via subscription   (Followers: 3)
Journal of Orthopaedics and Allied Sciences     Open Access   (Followers: 9)
Journal of Orthopaedics and Spine     Open Access   (Followers: 3)
Journal of Orthopaedics and Traumatology     Open Access   (Followers: 16)
Journal of Orthopaedics, Trauma and Rehabilitation     Open Access   (Followers: 6)
Journal of Orthopedics & Rheumatology     Open Access  
Journal of Orthopedics, Traumatology and Rehabilitation     Open Access   (Followers: 6)
Journal of Pediatric Orthopaedics     Hybrid Journal   (Followers: 15)
Journal of Prosthetics and Orthotics     Hybrid Journal   (Followers: 14)
Journal of Scleroderma and Related Disorders     Hybrid Journal  
Journal of the American Academy of Orthopaedic Surgeons     Hybrid Journal   (Followers: 12)
Journal of the American Podiatric Medical Association     Full-text available via subscription   (Followers: 8)
Journal of Traumatic Stress     Hybrid Journal   (Followers: 25)
Knee Surgery, Sports Traumatology, Arthroscopy     Hybrid Journal   (Followers: 27)
Multiple Sclerosis and Related Disorders     Hybrid Journal   (Followers: 8)
Musculoskeletal Care     Hybrid Journal   (Followers: 19)
Musculoskeletal Science and Practice     Hybrid Journal   (Followers: 3)
Nigerian Journal of Orthopaedics and Trauma     Open Access  
North American Spine Society Journal (NASSJ)     Open Access   (Followers: 3)
OA Orthopaedics     Open Access   (Followers: 7)
Obere Extremität     Hybrid Journal   (Followers: 1)
Open Journal of Orthopedics     Open Access   (Followers: 3)
Open Journal of Orthopedics and Rheumatology     Open Access  
Open Journal of Trauma     Open Access  
Open Orthopaedics Journal     Open Access  
Operative Orthopädie und Traumatologie     Hybrid Journal  
Operative Techniques in Orthopaedics     Full-text available via subscription   (Followers: 6)
Orthopädie & Rheuma     Full-text available via subscription  
Orthopädie und Unfallchirurgie up2date     Hybrid Journal  
Orthopaedic Journal of Sports Medicine     Open Access   (Followers: 14)
Orthopaedic Nursing     Hybrid Journal   (Followers: 11)
Orthopaedic Proceedings     Partially Free  
Orthopaedic Surgery     Open Access   (Followers: 1)
Orthopaedics & Traumatology: Surgery & Research     Full-text available via subscription   (Followers: 6)
Orthopaedics and Trauma     Full-text available via subscription   (Followers: 28)
Orthopedic Clinics of North America     Full-text available via subscription   (Followers: 5)
Orthopedic Research and Reviews     Open Access   (Followers: 6)
Orthopedic Reviews     Open Access   (Followers: 7)
Orthopedics     Full-text available via subscription   (Followers: 6)
Orthoplastic Surgery     Open Access  
Osteoarthritis and Cartilage     Full-text available via subscription   (Followers: 20)
Osteoarthritis and Cartilage Open     Open Access  
Osteologie     Hybrid Journal  
Osteoporosis and Sarcopenia     Open Access  
OTA International     Open Access  
Paediatric Orthopaedics and Related Sciences     Open Access   (Followers: 3)
Pain Management in General Practice     Full-text available via subscription   (Followers: 12)
Prosthetics and Orthotics International     Hybrid Journal   (Followers: 8)
Revista Brasileira de Ortopedia     Hybrid Journal  
Revista Chilena de Ortopedia y Traumatología / Chilean Journal of Orthopaedics and Traumatology     Open Access  
Revista Colombiana de Ortopedia y Traumatología     Full-text available via subscription  
Revista Cubana de Ortopedia y Traumatologí­a     Open Access  
Revista de la Asociación Argentina de Ortopedia y Traumatología     Open Access  
Revista Española de Cirugía Ortopédica y Traumatología     Full-text available via subscription   (Followers: 1)
Revista Portuguesa de Ortopedia e Traumatologia     Open Access  
Revue de Chirurgie Orthopédique et Traumatologique     Full-text available via subscription   (Followers: 3)
Romanian Journal of Orthopaedic Surgery and Traumatology     Open Access  
SA Orthopaedic Journal     Open Access   (Followers: 2)
SICOT-J     Open Access   (Followers: 1)
Spine     Hybrid Journal   (Followers: 73)
Spine Journal     Hybrid Journal   (Followers: 26)
Sport-Orthopädie - Sport-Traumatologie - Sports Orthopaedics and Traumatology     Full-text available via subscription   (Followers: 3)
Strategies in Trauma and Limb Reconstruction     Open Access   (Followers: 1)
Techniques in Orthopaedics     Hybrid Journal   (Followers: 6)
Therapeutic Advances in Musculoskeletal Disease     Hybrid Journal   (Followers: 5)
Trauma     Hybrid Journal   (Followers: 5)
Trauma (Travma)     Open Access  
Trauma und Berufskrankheit     Hybrid Journal  
Traumatology     Full-text available via subscription   (Followers: 1)
Traumatology and Orthopedics of Russia     Open Access  
Zeitschrift für Orthopädie und Unfallchirurgie     Hybrid Journal   (Followers: 2)
Ортопедия, травматология и протезирование     Open Access  

           

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  [1174 journals]
  • Prefabricated Trays Stock for Impression of Cleft lip and Palate
           Orthopedic Appliances: A Three-Dimensional Computational Analysis of
           Maxillary Jaws

    • Free pre-print version: Loading...

      Authors: Sara Sabir, Hasnaa Rokhssi, Noureddine Famma, Mohamed Azhari, Oussama Bentahar
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aimed to propose digitally designed impression trays, for newborns with cleft lip and palate (CLP) defects, using the computer-assisted design and manufacturing (CAD/CAM) technique, based on measurements of width, length, and height made on processed and scanned plaster models. Thus, we will describe the different software and concepts that can be used for the design of these impression trays.DesignA total of 59 plaster casts of newborn patients with CLP were collected and then scanned. After that, digital dental casts were three-dimensional (3D) evaluated, using precise anatomical landmarks.SettingThe Maxillofacial Prosthodontics Team at the Dental consultation and treatment center in Rabat, Morocco.Patients and ParticipantsThe study involved plaster casts of newborns with CLP, who had undergone presurgical neonatal treatment in the Maxillofacial Prosthodontics service in Rabat, Morocco.InterventionsNone,Main Outcome Measure(s)3D evaluation of 59 scanned plaster casts of newborn patients with CLP.ResultsThis work allowed us to obtain digital impression trays for each anatomical variation of orofacial clefts. These trays will limit the problems linked to conventional techniques for making neonatal orthopedic plates. We also emphasized the importance of a multidisciplinary approach involving several professionals in Morocco from various fields and specialties.ConclusionThis original technique is advantageous for the practitioner as well as for the patients and their parents. It allows for precise recording, better adaptation, time-saving, and parental comfort.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-10T05:21:53Z
      DOI: 10.1177/10556656221119075
       
  • Septal Lipoma and Cleft Palate: Particular Association, Particular
           Treatment'

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      Authors: Belmaati Cherkaoui Ghita, Souarji Ayyoub, Tajir Mariam, Oufkir Ayat Allah
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Congenital tumors of the oral cavity are infrequent and is dominated by teratogenic tumors. They are rarely associated with cleft palate. Congenital lipoma of the palate in patients with cleft palate remains exceptional. We report a case of congenital septal lipoma with cleft palate (the third case of such association is reported in the literature) and discuss the surgical treatment in light of the evolution of the patient.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-10T05:21:36Z
      DOI: 10.1177/10556656221117730
       
  • Spontaneous Reossification Following Craniectomy in a Pediatric Patient

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      Authors: Luke Soliman, Nikhil Sobti, Vinay Rao, Petra Klinge, Albert S. Woo
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Spontaneous reossification following a cranial defect is described by only a few case reports. A 6-month-old male with epidural hematoma underwent decompressive craniotomy, subsequently complicated by scalp abscess requiring removal of the bone flap. On serial outpatient follow-up, the patient demonstrated near-complete resolution of cranial defect over the course of 18 months, thus deferring the need for future cranioplasty. Prior articles have identified this occurrence in children and young adults; however, the present case is the first to report of this phenomenon in an infant less than 1 year of age. A brief review of the literature is provided with the proposed physiologic underpinning for the spontaneous reossification observed. While prior studies propose that recranialization is mediated by contact with the dura mater and pericranium, new investigations suggest that calvarial bone repair is also mediated by stem cells from the suture mesenchyme.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-03T04:08:40Z
      DOI: 10.1177/10556656221118426
       
  • Posttraumatic Growth and its Correlates Among Parents of Children With
           Cleft Lip and/or Palate

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      Authors: Miao He, Bing Shi, Qian Zheng, Caixia Gong, Hanyao Huang
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study examined the posttraumatic growth (PTG) about parents of children with cleft lip and/or palate (CL/P) and the correlates of PTG.DesignA cross-sectional study.Participants and SettingParents (N = 388) of children with isolated CL/P (ages 3 months-18 years) who had at least one cleft surgery within an oral and maxillofacial surgery department of a university-affiliated tertiary hospital in a provincial capital in southwest China.Main MeasuresDemographic information questionnaire, The Posttraumatic Growth Inventory, Simplified Coping Style Questionnaire, and Social Support Rating Scale were used for data collection.ResultsParents’ mean PTG score was in the moderate range (M = 65.7, SD = 13.73). PTG differed regarding the participants’ sex, ethnicity, and educational background. Pearson's correlation analysis revealed that positive coping (r = 0.43, P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-03T04:08:38Z
      DOI: 10.1177/10556656221118425
       
  • Anatomical Study of Domain Rescue of Palatal Length in Patients With a
           Wide Cleft Palate: Buccal Flap Reconstruction in Primary Palatoplasty

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      Authors: Kerry A. Morrison, Jenn Park, Danielle Rochlin, Margaret Lico, Roberto L. Flores
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundThis study characterizes the potential loss of velar length in patients with a wide cleft and rescue of this loss of domain by local flap reconstruction, providing anatomic evidence in support of primary lengthening of the soft palate during palatoplasty.MethodsA retrospective review was conducted of all patients with a cleft palate at least 10mm in width, who underwent primary palatoplasty with a buccal flap prior to 18 months of age over a 2-year period. All patients underwent primary palatoplasty with horizontal transection of the nasal mucosa, which was performed after nasal mucosa repair, but prior to muscular reconstruction. The resulting palatal lengthening was measured and the mucosal defect was reconstructed with a buccal flap.ResultsOf the 22 patients included, 3 (13.6%) had a history of Pierre Robin sequence, and 5 (22.7%) had an associated syndrome. No patients had a Veau I cleft, 7 (31.8%) had a Veau II, 12 (54.5%) had a Veau III, and 3 had (13.6%) a Veau IV cleft. All patients had a right buccal flap during primary palatoplasty. The mean cleft width at the posterior nasal spine was 10.6  ±  2.82mm, and mean lengthening of the velum after horizontal transection of the nasal mucosa closure was 10.5  ±  2.23mm. There were 2 (9.1%) fistulas, 1 (4.5%) wound dehiscence, 1 (4.5%) 30-day readmission, and no bleeding complications.ConclusionsPatients with a wide cleft palate have a potential loss of 1cm velar length. The buccal flap can rescue the loss of domain in palatal length, and potentially improve palatal excursion.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-03T04:08:33Z
      DOI: 10.1177/10556656221117930
       
  • Is the Nasolabial Region Symmetric in Individuals With Cleft Lip and
           Palate'

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      Authors: Furkan Dindaroglu, Ege Dogan, Servet Dogan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo assess the nasolabial symmetry in smile and resting position in individuals with nonsyndromic unilateral cleft lip and palate (UCLP) and compare with Class I individuals without cleft lip and palate (CLP).Materials and MethodsFrontal photographs of 40 individuals with UCLP (mean age 14.30 ± 0.25 years; 20 female, 20 male) and 40 skeletal Class I individuals without CLP (mean age 14.60 ± 0.34 years; 20 female and 20 male) were examined. Bland–Altman Levels of Agreements, Mann–Whitney U test, independent sample t-test were conducted, and intraclass correlation coefficients (ICCs) were calculated for statistical analysis.ResultsDifferences in the transverse lip distance (d-d’) and vertical lip height at crista philtri (e-e’) were lesser in the UCLP group at smile (P = .001; P = .005, respectively). When comparing the differences in these measurements at rest between control and UCLP groups, vertical lip height was longer at crista philtri (e-e’) (P = .014), crista philtri was more laterally located (f-f’) (P = .002), and the transverse lip distance was greater (d-d’) (P = .004) in the UCLP group. Distance of the crista philtri to the midline of the face (f-f’) (P = .007) were higher in the UCLP group during smiling compared to control group.ConclusionsNasolabial region asymmetry in smiling and rest position in individuals with UCLP was significantly different from the control group. However, the asymmetry in the nasolabial region, which is more evident in the resting position in individuals with UCLP, decreases during smiling.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-01T07:37:01Z
      DOI: 10.1177/10556656221116535
       
  • Prevalence and Features of Maxillary Sinus Septa in Patients with Cleft
           Lip and Palate: Cone Beam Computed Tomography Imaging Technique

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      Authors: Maryam Paknahad, Samira Zahedrozegar, Neda Babanouri, Shabnam Ajami
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe development of the maxillary sinus is different in patients with cleft lip and palate (CLP) compared to non-CLP individuals. To investigate the prevalence and features of maxillary sinus septa (MSS) in patients with CLP in comparison with the non-CLP population.DesignRetrospective study.InterventionCone beam computed tomography (CBCT) evaluation.SettingCLP center in Shiraz faculty of dentistry, Iran.PatientsA total 306 sinuses (88 cleft and 218 noncleft) on 153 images (CLP group: n  =  66; control group: n  =  87) were examined to determine the prevalence of septa and characterize them.Main Outcome MeasuresSinus septa were characterized according to height, orientation, angle, origin, and location. The chi-square test, Mann-Whitney U test, and Fisher's exact test were used for statistical analysis.ResultsThe prevalence of septa was 28.9% and 32.1% in the CLP and control groups, respectively. No significant difference was found between the study groups in terms of prevalence, location, and orientation of MSS. The average height and angle of septa were significantly higher in the control group compared to the CLP group. Inferior origin was significantly more prevalent in the control group than in the CLP group (P = .004).ConclusionThere was no difference in the prevalence of MSS between patients with CLP and non-CLP individuals. However, certain features of the septa were different in patients with CLP.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-01T07:36:51Z
      DOI: 10.1177/10556656221117732
       
  • Impact of a Palatal Fistula After Cleft Palate Repair on Velopharyngeal
           Closure

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      Authors: Mackenzie M. Geisman, Gary B. Skolnick, Lynn M. Grames, Sybill D. Naidoo, Alison K. Snyder-Warwick, Kamlesh B. Patel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      PurposeA palatal fistula is an adverse outcome of cleft palate repair. It is unknown if a palatal fistula will influence velopharyngeal closure, even after repair of the fistula. This study determines the effect of a soft palate fistula on the risk of developing velopharyngeal insufficiency.MethodsA retrospective chart review was conducted on patients who underwent primary cleft palate repair between 2000 and 2015, with complete records at 4 years of age. Fistulae involving the secondary palate following primary palatoplasty were classified as the soft or hard palate. A forced-entry multivariate logistic regression model was built to detect predictors of velopharyngeal dysfunction.ResultsRecords of 329 patients were analyzed with a mean follow-up of 8.7 years. A palatal fistula was identified in 89/329 patients (27%) and 29/329 patients (9%) underwent an independent fistula repair. Of the patients with fistula, 44% were located in the hard palate only and 56% had soft palate involvement. Compared to patients without a fistula, rates of velopharyngeal dysfunction were significantly higher in patients with a fistula involving the soft palate (OR 3.875, CI: 1.964-7.648, P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-01T07:36:41Z
      DOI: 10.1177/10556656221116534
       
  • Are Children with Cleft Palate at Increased Risk for Laryngeal
           Pathology'

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      Authors: Robert Brinton Fujiki, Susan L. Thibeault
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:To determine the prevalence of laryngeal pathology in children presenting with cleft palate with or without cleft lip (CP ± L) who underwent nasoendoscopy to assess palatal function. A secondary aim was to determine the relationship between patient demographics, resonance, articulation, and prevalence of laryngeal pathology in this population.Design:Retrospective, observational cohort studySetting:Outpatient pediatric cranio-facial anomalies clinicPatients:Children ≤18 years of age presenting with CP ± L (N = 215) who underwent nasoendoscopy, speech language pathology, plastic surgery, and otolaryngological evaluations between 2009 and 2020.Main Outcome Measure:Laryngeal diagnosis by pediatric otolaryngologists.Results:21.9% of children presented with laryngeal pathology. Diagnoses included benign vocal fold lesions and laryngeal edema sufficiently severe to alter vocal fold edge contour. Likelihood of laryngeal pathology increased by approximately 12% with every increase of 1 year in age (P = .001, OR = 1.12). Children with laryngeal pathology were 50% more likely to have undergone palatal repair (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-08-01T07:36:31Z
      DOI: 10.1177/10556656221104027
       
  • Cleft Lip and/or Palate Repair in Children With Hypopituitarism: Analysis
           of the Kids’ Inpatient Database

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      Authors: Nicolas S. Poupore, Shreya Chidarala, Shaun A. Nguyen, Ronald J. Teufel, Krishna G. Patel, Phayvanh P. Pecha, William W. Carroll
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveChildren with hypopituitarism (CwHP) can present with orofacial clefting, frequently in the setting of multiple midline anomalies. Hypopituitarism (HP) can complicate medical and surgical care; the perioperative risk in CwHP during the traditionally lower risk cleft lip and/or palate (CL/P) repair is not well described. The objective of this study is to examine the differences in complications and mortality of CL/P repair in CwHP compared to children without hypopituitarism (CwoHP).DesignA retrospective cross-sectional analysis.SettingThe 1997 to 2019 Kids’ Inpatient Databases (KID).PatientsChildren 3 years old and younger who underwent CL/P repair.Main Outcome Measure(s)Complications and mortality.ResultsA total of 34 106 weighted cases were analyzed, with 86 having HP. CwHP had a longer length of stay (3.0 days [IQR 2.0-10.0] vs 1.0 day [IQR 1.0-2.0], P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-29T07:28:56Z
      DOI: 10.1177/10556656221117435
       
  • Optimizing Blood Loss and Management in Craniosynostosis Surgery: A
           Systematic Review of Outcomes Over the Last 40 Years

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      Authors: Demetrius M. Coombs, Rebecca Knackstedt, Niyant Patel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveSurgical correction of craniosynostosis can involve significant blood loss. Rates of allogenic blood transfusion have been reported to approach 100%. Multiple interventions have been described to reduce blood loss and transfusion requirements. The aim of this study was to analyze various approaches over the last 4 decades to optimize blood loss and management during craniosynostosis surgery.DesignPRISMA guidelines for systematic reviews were followed. PubMed and Cochrane database searches identified studies analyzing approaches to minimizing blood loss or transfusion rate in craniosynostosis surgery.Main Outcome MeasuresPrimary outcomes included rate or amount of allogenic or autologous blood transfusion, estimated blood loss (EBL), postoperative hemoglobin (Hg), or hematocrit (Hct) levels. Secondary outcomes were examined when reported.ResultsFifty-two studies met inclusion criteria. There was marked heterogeneity regarding design, inclusion criteria, surgical intervention, and endpoints. The majority of the studies were nonrandomized and noncomparative. Four studies analyzed erythropoietin (EPO), 6 analyzed various cell-saver (CS) technologies, 18 analyzed antifibrinolytics (tranexamic acid [TXA], aminocaproic acid [ACA], and aprotinin [APO]), 8 analyzed various alternatives, and 16 analyzed multimodal pathways & protocols. Some studies analyzed multiple approaches.ConclusionsAlthough the majority of studies reviewed represent level III/IV evidence, several high-quality level I studies were identified and included. Level I evidence supported an improvement in blood outcomes by utilizing EPO, CS, and TXA, individually or in concert with one another. Thus, this review suggests that a multi-prong approach may be the most effective means to optimize blood loss and transfusion outcomes in craniosynostosis surgery.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-29T06:50:56Z
      DOI: 10.1177/10556656221116007
       
  • Intellectual Functioning of Children With Isolated PRS, PRS-Plus, and
           Syndromic PRS

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      Authors: Stephanie Malarbi, Anita K. Chisholm, Julia K. Gunn-Charlton, Alice C. Burnett, Tiong Yang Tan, Shirley S. W. Cheng, Anastasia Pellicano, Jocelyn Shand, Andrew Heggie, Rod W. Hunt
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveDescribe the intelligence quotient (IQ) of children with Pierre Robin sequence (PRS).DesignProspective cohort study.SettingNeurodevelopmental follow-up clinic within a hospital.PatientsChildren with PRS (n = 45) who had been in the Neonatal Intensive Care Unit (NICU) were classified by a geneticist into 3 subgroups of isolated PRS (n = 20), PRS-plus additional medical features (n = 8), and syndromic PRS (n = 17) based on medical record review and genetic testing.Main Outcome MeasureChildren with PRS completed IQ testing at 5 or 8 years of age with the Wechsler Preschool and Primary Scale of Intelligence, Third Edition (WPPSI-III) or Fourth Edition (WPPSI-IV) or the Wechsler Intelligence Scale for Children, Fourth Edition (WISC-IV) or Fifth Edition (WISC-V).ResultsIQ scores were more than 1 to 2 standard deviations below the mean for 36% of the overall sample, which was significantly greater compared to test norms (binomial test P = .001). There was a significant association between PRS subtype and IQ (Fisher’s exact P = .026). While only 20% of children with isolated PRS were within 1 standard deviation below average and 35% of children with syndromic PRS were below 1 to 2 standard deviations, 75% of PRS-plus children scored lower than 1 to 2 standard deviations below the mean.ConclusionPRS subgroups can help identify children at risk for cognitive delay. The majority of children with PRS-plus had low intellectual functioning, in contrast to the third of children with syndromic PRS who had low IQ and the majority of children with isolated PRS who had average or higher IQ.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-28T05:51:17Z
      DOI: 10.1177/10556656221115596
       
  • Premaxillary Setback With Posterior Vomerine Ostectomy and Complete
           Bilateral Cleft Lip Repair: Report of a Case With Challenging Anatomy

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      Authors: Usama S. Hamdan, Antonio M. Melhem, Mario Haddad, Robert Anthony Younan, Bonnie Padwa, Rami Kantar
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Congenital clefts of the lip and/or palate are among the most common craniofacial malformations. Patients with bilateral cleft of the lip often present with projected premaxilla. Premaxillary setback with a vomerine ostectomy posterior to the vomero-premaxillary suture, bilateral cleft lip repair, bilateral gingivoperiosteoplasties, and primary cleft lip rhinoplasty are achieved in a single-stage surgery that provides a valuable alternative to patients, especially in the outreach settings. In this article, we present a case report of a patient born with a bilateral cleft of the lip and a protruded premaxilla. He had collapsed secondary palatine shelves requiring intraoperative manual expansion to ensure access to the vomer bone.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-28T05:51:08Z
      DOI: 10.1177/10556656221104028
       
  • Screening for Academic Risk Among Students With Cleft Lip and/or Palate:
           Patterns of Risk and Qualities of Effective Tools

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      Authors: Amy L. Conrad, Claudia Crilly Bellucci, Celia E. Heppner, Meredith Albert, Canice E. Crerand, Suzanne Woodard, Farah Sheikh, Kathleen A. Kapp-Simon
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study evaluated the effectiveness of academic screening measures in relation to parent-reported diagnoses.DesignMulticenter, retrospective cohort study including structured interviews, questionnaires, and chart reviews.SettingSix North American cleft centers.ParticipantsChildren (n = 391) with cleft lip and/or palate, ages 8 to 10 years of age (192 male) and their guardians were recruited during regular clinic visits.Main Outcome MeasuresParent and child ratings on the Pediatric Quality of Life Inventory (PedsQL) School Scale, child report on CleftQ School Scale, parent report on the Adaptive Behavior Assessment System-Third Edition Functional Academics (ABAS-FA) Scale and Child Behavior Checklist (CBCL) School Competency Scale, parent interview, and medical chart review.ResultsRisk for concerns ranged from 12% to 41%, with higher risk reflected on the CBCL-SC compared to other measures. Males with cleft palate were consistently at the highest risk. Only 9% of the sample had a parent-reported diagnosis of a learning or language disability. Ratings from the ABAS-FA and CBCL-SC had the highest utility in identifying those with language and/or learning concerns.ConclusionsAs cleft teams work to develop standardized batteries for screening and monitoring of patients, it is important to evaluate the effectiveness of measures in identifying those at highest risk. When screening for language and learning disorders, questions related to potential academic struggles, such as increased school effort or increased school distress, are most useful. Referrals for follow-up evaluation are recommended for those identified at high risk.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-27T06:00:59Z
      DOI: 10.1177/10556656221116008
       
  • A Rare Case of Cleft Palate Associated With Tongue Hamartoma: A Case
           Report and Systematic Review

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      Authors: Suvashis Dash, Shashank Chauhan, Karthik Sennimalai, Om Prakash Kharbanda, Maneesh Singhal
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionPalate development involves a genetic regulation through a complex molecular mechanism that may be disrupted by environmental factors, resulting in impaired fusion and cleft palate formation. An encounter with a case of cleft palate due to dorsal tongue hamartoma prompted us to perform this systematic review.ObjectiveTo review the clinical profile and management approach for a case with cleft palate and tongue hamartoma.DesignA systematic literature search was conducted using keywords related to cleft palate and tongue hamartoma in PubMed, Scopus, MEDLINE, and Scielo databases through December 2021, with no time or language restrictions.Patients, ParticipantsStudies reporting patients with cleft palate and tongue hamartoma were included.Main outcome measure(s)Information related to clinical profile, diagnostic tests, histopathology, management, and outcomes were extracted.ResultsFourteen relevant publications were identified with 16 cases reported so far. Among them, thirteen patients were females (81.25%), and 3 were males (18.75%). The age of presentation varied from birth to 19 years. Oral-facial-digital syndrome (type II) was the most commonly associated syndrome.ConclusionCongenital tongue hamartoma with cleft palate is a rare presentation, which can present as an isolated entity or part of a syndrome. Genetic evaluation is warranted, particularly for multiple hamartomatous lesions. The preferred treatment is immediate excision of hamartoma while following a standard timeline for palatoplasty.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-26T04:06:47Z
      DOI: 10.1177/10556656221116001
       
  • Sagittal Growth Restriction of the Midface Following Isolated Cleft Lip
           Repair: A Systematic Review and Meta-Analysis

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      Authors: Karel-Bart Celie, Jordan Wlodarczyk, Priyanka Naidu, Maria Fernanda Tapia, Eric Nagengast, Caroline Yao, William Magee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveMidface hypoplasia (MFH) is a long-term sequela of cleft lip and palate repair, and is poorly understood. No study has examined the aggregate data on sagittal growth restriction of the midface following repair of the lip, but not palate, in these patients.MethodsA systematic review of 3780 articles was performed. Twenty-four studies met inclusion criteria and 11 reported cephalometric measurements amenable to meta-analysis. Patients with Veau class I-III palatal clefts were included so long as they had undergone only lip repair. Groups were compared against both noncleft and unrepaired controls.ResultsCephalometrics were reported for 326 patients (31.3% female). Noncleft controls had an average SNA angle of 81.25° ± 3.12°. The only patients demonstrating hypoplastic SNA angles were those with unilateral CLP with isolated lip repair (77.4° ± 4.22°). Patients with repaired CL had SNA angles similar to noncleft controls (81.4° ± 4.02°). Patients with unrepaired CLP and CL tended toward more protruding maxillae, with SNA angles of 83.3° ± 4.04° and 87.9° ± 3.11°, respectively. Notably, when comparing SNA angles between groups, patients with CLP with isolated lip repair had significantly more hypoplastic angles compared to those with repaired CL (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-25T11:34:10Z
      DOI: 10.1177/10556656221116005
       
  • Does YouTubeTM Offer High-Quality Information About Nasoalveolar
           Molding'

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      Authors: Can Arslan, Elif Ceren Aksahin, Rahime Burcu Nur Yılmaz, Derya Germec Cakan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo assess the content and quality of YouTubeTM videos concerning nasoalveolar molding (NAM).DesignYouTubeTM was searched for videos containing information relevant to NAM with the 2 keywords “nasoalveolar molding,” and “presurgical infant orthopedics.” A total of 24 out of 51 videos were found to be applicable to this study and rated for quality using the Global Quality Scale (GQS). To determine whether the contents of the selected 24 videos were useful or not, a content usefulness index consisting of 8 parameters was created. The videos were classified according to the usefulness index as low or high content videos. Spearman rank correlation analysis, Kolmogorov-Smirnov, Shapiro-Wilk, and Mann-Whitney U-tests were used for statistical analysis.ResultsThe mean GQS score of the 24 YouTubeTM videos on NAM was 2.3 ± 0.8, indicating overall poor quality. In terms of information, videos with high content (29.2%) were less in number than low content videos (70.8%). GQS values were found to be significantly higher in the high content group (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-21T03:09:18Z
      DOI: 10.1177/10556656221115025
       
  • Relationship Between Stigma Experience and Self-Perception Related to
           

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      Authors: Ayaka Oka, Chihiro Tanikawa, Hauka Ohara, Takashi Yamashiro
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo examine the relationship between stigma experience related to facial appearance in Japanese youths with cleft lip and/or palate (CL/P) and their self-perception.DesignA cross-sectional study.ParticipantsSixty-nine Japanese youths with CL/P (11-18 years old)Outcome measuresThe participants’ stigma experience in relation to facial appearance (measured with 7 single contextual scale items) and their self-perception (measured with 5 domain scores based on 30 perceptual items) were assessed using the Japanese version of the Youth Quality of Life Instrument-Facial Differences Module. Participants were categorized into high and low self-perception subgroups with a threshold of 1 standard deviation for each domain. The frequency of stigma experiences was compared between the following 2 subgroups: age, sex, cleft palate only versus other cleft, and high versus low self-perception. Correlations between the responses regarding stigma and all domain scores were examined.ResultsSixteen percent of the participants reported experiencing stigma. Hearing others say something about their face occurred significantly more frequently in youths 15 to 18 years of age than in youths 11 to 14 years of age. Stigma frequency was not found to differ by sex or cleft type. Stigma experiences were significantly more frequent for youth with higher scores across negative self-perception domains as well as higher coping skills. Significant correlations were identified between responses regarding stigma items and all domain scores (r  =  0.27-0.63, p 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-21T03:08:10Z
      DOI: 10.1177/10556656221114581
       
  • Review of Diet Protocols Following Orthognathic Surgery and Analysis of
           Postoperative Weight Loss

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      Authors: Zhazira Irgebay, Jamison C. Beiriger, Justin W. Beiriger, Sayna Matinrazm, Megan Natali, Cleo Yi, John Smetona, Lindsay Schuster, Jesse A. Goldstein
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionOrthognathic surgery is routinely practiced, yet little comparative data exists to evaluate post-orthognathic surgery diet protocols.ObjectiveTo evaluate which postoperative diet protocols are recommended and to quantify post-orthognathic surgery weight changes in our institutional cohort.MethodsAn internet search was carried out on Google for “orthognathic surgery diet” and the postoperative diet recommendations from centers worldwide were quantified. Additionally, a retrospective analysis of patients that underwent orthognathic surgery at our institution was performed, and their preoperative and postoperative weights were recorded.ResultsThe internet search yielded 58 centers that met our inclusion criteria. Most centers were in the United States (n = 37, 63.8%) and were oral and maxillofacial surgeon (OMFS)-led centers (n = 39, 67.2%). Postoperative diets were categorized into 7 distinct protocols, ranging from most to least restrictive—the most popular was liquid diet for 2 to 4 weeks followed by soft diet for 2 to 6 weeks. There were no significant patterns observed across different geographical regions or specialties.In our institution, 135 patients were identified. Overall, there was an average maximum weight loss of 4.1 kg by week 4, followed by a gradual increase in weight. Linear regression analysis showed that patients with greater preoperative body mass index (BMI) lost more weight postoperatively than patients with lower BMI (R2 = 0.25, P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-15T07:10:37Z
      DOI: 10.1177/10556656221113998
       
  • “Septoplasty” Performed at Primary Cleft Rhinoplasty: A Systematic
           Review of Techniques and Call for Accurate Terminology

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      Authors: Jenn J. Park, Ricardo Rodriguez Colon, Fernando D. Arias, Matteo Laspro, Bachar F. Chaya, Danielle H. Rochlin, David A. Staffenberg, Roberto L. Flores
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivePrimary cleft nasal repair can include septal reconstruction. We hypothesize that primary cleft septoplasty and adult septoplasty have fundamental differences that render these procedures as distinct surgical entities.DesignSystematic review of the PubMed, Cochrane, and Embase databases performed on pediatric cleft and general adult septoplasty techniques through December 2021. (PROSPERO ID CRD42022295763)Main Outcome MeasuresCollected data included information on septal dissection, septal detachment, and management of the bony and cartilaginous septum.ResultsTwenty-eight pediatric cleft septoplasty and 229 adult septoplasty studies were included. Dissection in primary cleft septoplasty was limited to the anterocaudal septum, while secondary cleft septoplasty and adult septoplasty techniques entailed wide exposures of the cartilaginous septum with or without exposure of the perpendicular plate of the ethmoid. In primary cleft septoplasty, detachment of the septum was mostly limited to the nasal spine and anterior base of the cartilaginous septum, while secondary cleft septoplasty and adult septoplasty included detachment from the vomer, and ethmoid. In the few reports of cartilage excision during primary cleft septoplasty, removal was limited to the anterior inferior border of the septum, while secondary cleft septoplasty and adult septoplasty included excision of the cartilaginous and bony septum.ConclusionPrimary cleft septoplasty is distinct from septoplasty performed on facially mature patients. More specifically, septal dissection and detachment are limited to the anterior caudal area during primary lip repair, with rare removal of cartilage or bone. Given these differences, the authors suggest the term “septal reset” to describe septoplasty performed during primary cleft nasal repair.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-15T07:10:25Z
      DOI: 10.1177/10556656221113997
       
  • Denosumab Therapy in Cherubism

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      Authors: Steele I. Liles, Ian C. Hoppe, Laura Arnold
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Cherubism is a rare disorder characterized by proliferative fibro-osseous lesions that result in bilateral bony hyperplasia of the face. Management varies based on symptom severity and includes longitudinal follow-up, pharmacotherapy, and/or surgical debulking. Off-label treatment with denosumab, a human monoclonal antibody that binds RANKL and inhibits osteoclast function to reduce bone resorption, can be beneficial in suppressing the proliferation of bone to minimize the need for surgery and to control postoperative reproliferation. Close follow-up is needed to maintain appropriate electrolyte levels. The present case demonstrates the achievement of symptomatic control with denosumab in a child with severe refractory cherubism.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-13T03:54:18Z
      DOI: 10.1177/10556656221113891
       
  • Is the Message Clear' Evaluation of Readability Levels for Cleft Lip,
           Cleft Palate, and Craniofacial Websites

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      Authors: George N. Washington, Alfredo Cepeda, Joseph Moffitt, Connor K Groff, Matthew R. Greives, Phuong D. Nguyen
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveWeb-based health information is the leading source of medical knowledge for patients and families. The American Medical Association (AMA) and US Department of Health and Human Services recommend reading material be at or below a sixth-grade reading level. This study aimed to evaluate and compare the readability of the most popularly searched cleft lip and/or palate (CL/P) and other craniofacial syndrome (CFS) websites.DesignGoogle searches for “cleft lip,” “cleft palate,” and “craniofacial syndromes” were performed to identify the top 40 websites in an incognito window with the location set to the United States. Flesch Reading Ease Score (FRES) was used to determine ease of reading from 0 (most difficult) to 100 (greatest ease of reading) and Flesch-Kincaid Reading Grade (FKGL) for website content and compared between websites using an FRES of 80 to 90 and FKGL of 6.0 to 6.9 for a sixth-grade reading level.ResultsReadability was low for all sites with 6 CL/P websites and no CFS websites at or below a sixth-grade reading level. CL/P websites had FRES readability scores of 58.5 ± 12.3 and were at a 9.4 ± 2.3 grade level. CFS websites had readability scores on the FRES of 39.8 ± 13.1 and were at a 10.8 ± 1.8 grade level.ConclusionsWeb-based information related to CL/P and CFS is on average several grade levels above the recommended sixth-grade reading level. Online information for CL/P and CFS may need to be revised to improve understanding for the public and families.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-05T06:23:04Z
      DOI: 10.1177/10556656221112672
       
  • A Systematic Review of the Cost-Effectiveness of Cleft Care in Low- and
           Middle-Income Countries: What is Needed'

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      Authors: Karen Y. Chung, George Ho, Aysegul Erman, Joanna M. Bielecki, Christopher R. Forrest, Beate Sander
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe objective of this paper is to conduct a systematic review that summarizes the cost-effectiveness of cleft lip and/or palate (CL/P) care in low- and middle-income countries (LMICs) based on existing literature.DesignWe searched eleven electronic databases for articles from January 1, 2000 to December 29, 2020. This study is registered in PROSPERO (CRD42020148402). Two reviewers independently conducted primary and secondary screening, and data extraction.SettingAll CL/P cost-effectiveness analyses in LMIC settings.Patients, ParticipantsIn total, 2883 citations were screened. Eleven articles encompassing 1,001,675 patients from 86 LMICs were included.Main Outcome MeasuresWe used cost-effectiveness thresholds of 1% to 51% of a country's gross domestic product per capita (GDP/capita), a conservative threshold recommended for LMICs. Quality appraisal was conducted using the Joanna Briggs Institute (JBI) checklist.ResultsPrimary CL/P repair was cost-effective at the threshold of 51% of a country's GDP/capita across all studies. However, only 1 study met at least 70% of the JBI criteria. There is a need for context-specific cost and health outcome data for primary CL/P repair, complications, and existing multidisciplinary management in LMICs.ConclusionsExisting economic evaluations suggest primary CL/P repair is cost-effective, however context-specific local data will make future cost-effectiveness analyses more relevant to local decision-makers and lead to better-informed resource allocation decisions in LMICs.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-04T05:34:45Z
      DOI: 10.1177/10556656221111028
       
  • The Esthetic Perception of Morphological Severity in Scaphocephalic
           Patients is Correlated With Specific Head Geometrical Features

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      Authors: Alessandro Borghi, P. Heutinck, N. Rodriguez-Florez, M. Koudstaal, F. Ruggiero, S. Ajami, S. Schievano, N.U.O. Jeelani, D. Dunaway
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo investigate the relationship between perception of craniofacial deformity, geometric head features, and 3D head shape analyzed by statistical shape modeling (SSM).PatientsA total of 18 unoperated patients with scaphocephaly (age  =  5.2  ±  1.1m)—6 were followed-up after spring-assisted cranioplasty (SAC) (age  =  9.6  ±  1.5m)—and 6 controls (age  =  6.7  ±  2.5m).Main Outcome Measures3D head shapes were retrieved from 3D scans or computed tomography (CTs). Various geometrical features were measured: anterior and posterior prominence, take-off angle, average anterior and posterior lateral and horizontal curvatures, cranial index (CI) (cranial width over length), and turricephaly index (TI) (cranial height over length). SSM and principal component analysis (PCA) described shape variability. All models were 3D printed; the perception of deformity was blindly scored by 9 surgeons and 1 radiologist in terms of frontal bossing (FB), occipital bulleting (OB), biparietal narrowing (BN), low posterior vertex (LPV), and overall head shape (OHS).ResultsA moderate correlation was found between FB and anterior prominence (r  =  0.56, P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-04T05:34:16Z
      DOI: 10.1177/10556656221111307
       
  • Raising a Child with Craniosynostosis: Psychosocial Adjustment in Parents

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      Authors: Bruna Costa, Wendy Edwards, Laura Culshaw, Karen Wilkinson-Bell, Nicola Marie Stock
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveWhile knowledge of the psychosocial impact of craniofacial conditions is growing, literature regarding the impact on parents remains limited. Parents of children born with a health condition may be at risk of experiencing a range of psychosocial challenges. This study conducted an initial investigation of psychosocial adjustment of parents of individuals with craniosynostosis to inform support provision for families.SettingAn online UK-wide mixed-methods survey was distributed to members of Headlines Craniofacial Support.DesignQuantitative data including standardized measures were analysed using descriptive statistics and independent samples t-tests, and inductive content analysis was used for open-ended questions.ParticipantsMothers (n  =  109) and fathers (n  =  9) of individuals ages 3 months to 49 years with single suture (63%) or syndromic (33%) craniosynostosis participated.ResultsCompared to the general population, parents of individuals with craniosynostosis reported higher levels of stress, anxiety, and depression; lower levels of resilience and optimism. Qualitative responses provided insight into parents’ experiences of birth, diagnosis, healthcare provision, familial wellbeing, and relationships. Parents reported several unmet information and support needs, alongside a range of positive outcomes.ConclusionsThis study illustrates the potential long-term psychosocial implications for parents raising children with craniosynostosis. There is a need for routine psychological screening for family members and provision of appropriate psychological support for those at risk for distress. Non-specialist health professionals may benefit from additional training about craniofacial conditions so they are better equipped to support and refer families.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-07-04T05:30:53Z
      DOI: 10.1177/10556656221102043
       
  • Arch Symmetry in Patients Without and With Cleft Lip and Palate After
           Orthodontic/Rehabilitative Treatment—A Stereophotogrammetry Study

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      Authors: Jefferson Freire Cardoso, Maria Giulia Rezende Pucciarelli, José Antonio Siqueira Laurenti, Andreia Fernandes Emilio Laposta, Karin Hermana Neppelenbroek, Thaís Marchini Oliveira, Simone Soares
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo evaluate and compare the dental arch symmetry of individuals with and without cleft lip and palate after orthodontic/rehabilitation treatment.DesignCross-sectional study.SettingTertiary cleft center in Brazil.PatientsFifty-five participants aged between 18 and 30 years were divided into 3 groups according to treatment. Patients that received either a fixed partial denture (FPD) or implant-supported crown (ISC) in the cleft area or only orthodontic treatment, noncleft patients (NC).InterventionsAn analysis was performed using digitized dental casts scanned by laser and software. The following linear measurements were evaluated: incisor-canine; canine-molar; incisor-molar; surface and volume of the palatal region.Main Outcome MeasureThree-way ANOVA was used to compare the study factors: group (FPD/ISC/NC) and side (right/left) followed by the Tukey test to verify their interaction (α = .05).ResultsThe results showed statistically significant differences among groups for the maxillary linear measurements canine-molar and incisor-molar, but not for incisor-canine. No statistically significant differences were found regarding the side for the maxillary measurements, while the factor interaction showed similarity only for incisor-canine. The mandibular measurements showed no statistical differences among groups, sides, or factor interactions. In surface and volume, all values in patients with cleft presented lesser than in without cleft patients.ConclusionRegardless of the rehabilitation, arch symmetry can be achieved in the incisor-canine dimension in the cleft area.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-30T06:15:10Z
      DOI: 10.1177/10556656221110096
       
  • Reconstruction of the Nose After Unilateral Cheilouranoplasty

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      Authors: Vladimir A. Vissarionov, Magomet Sh. Mustafaev, Sofiyat M. Mustafaeva, Irina A. Karyakina, Dzhambulat T. Kuzhonov, Muslim Sh. Mustafaev
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveCongenital clefts of the maxillofacial area still remain of current interest in reconstructive facial surgery. While their frequency grows up, the issues of effective surgical primary and secondary interventions are not completely solved yet. The article presents the main problems associated with the elimination of the deformations and our modifications of methods for their correction.SettingThe study was conducted based on the Centre [2] and the University [1], Russian Federation. The project was carried out within the framework of the state assignment of the Ministry of Science and Higher Education of the Russian Federation, mnemocode 0669-2020-0008.Patients, ParticipantsThe research is based on the results of complex treatment of 112 patients with unilateral clefts of the upper lip and palate from the age of 5 to 34 years, 68 patients of them also carried out secondary surgeries.InterventionsThe methods of performing rhinocheiloplasty by moving a “sliding” flap, of eliminating a ctenoid plica appearing after primary surgeries are presented, described, explained, and substantiated in the article.ResultsThe proposed methods gave a good or satisfactory result in 92.8% of the operated patients, which allows them to be considered effective for eliminating congenital and postoperative nasal deformities in patients with facial clefts.Conclusions: The surgical methods of eliminating congenital and postoperative deformities of the nose in patients with facial clefts should be modified in accordance with the nature of the most frequently arising shortcomings of the surgical stages of correction.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-30T06:14:04Z
      DOI: 10.1177/10556656221099816
       
  • Success in Emergency Treatment of Neonatal Giant Teratoma with Cleft
           Palate: A Case Report

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      Authors: Shengyou Ge, Ning Wang, Wei Shang, Yuanyong Feng
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Neonatal teratoma, a common congenital malformation, rarely occurs in the head and neck region, especially not within the oral cavity. This report presents a case of neonatal giant teratoma in the oral cavity and oropharynx along with cleft palate, which caused postnatal airway obstruction and respiratory distress and required postnatal resection in a female newborn. After the delivery and routine neonatal examination, the anesthesiologist conducted orotracheal intubation to establish the airway, and tumor resection was immediately done under local anesthesia. The optimal treatment of neonatal teratoma is exclusive emergent surgery. Immediate postnatal resection is necessary to prevent airway obstruction.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-24T05:20:02Z
      DOI: 10.1177/10556656221109753
       
  • Prognostic Factors for Orthognathic Surgery in Children With Cleft Lip
           and/or Palate: Dentition and Palatal Morphology

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      Authors: Jaeyeon Lim, Chihiro Tanikawa, Mikihiko Kogo, Takashi Yamashiro
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo determine dental and palatal morphology in children with cleft lip and/or palate (CL/P) and identify morphological prognostic factors for orthognathic surgery (OGS).DesignRetrospective cohort study.SettingOrthodontic department of a university dental hospital.ParticipantsThis study included 80 patients with bilateral and unilateral CL/P who had lateral cephalograms at the ages of 7 (T1), 15 (T2) years, and a dental plaster model at T1.Main Outcome MeasuresPlaster models at T1 were scanned with a three-dimensional (3D) scanner. Morphological features were extracted from 3D models with geometric morphometrics software as principal components (PCs). The combinations of the PCs and other predictive factors (ie, the No. of clefts in the lip and alveolus, the palatal repair method, sex, cephalometric variables at T1, and the No. of missing teeth) were examined by logistic regression to determine the predictability for OGS. The need for OGS and skeletal and dental discrepancies at T2 were examined as outcomes.ResultsShrinkage of the palate, including vertical shallowing and transverse narrowing of the posterior maxilla and cleft-side asymmetry of the anterior maxilla at T1, as well as the No. of clefts in the lip and alveolus, the palatal repair method, male sex, several cephalometric variables for the sagittal and vertical dimensions, and the No. of missing teeth, were found to be predictive factors for OGS.ConclusionsMorphological prognostic factors for OGS in children with CL/P were determined.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-24T01:38:41Z
      DOI: 10.1177/10556656221109425
       
  • Investigating the Impact of Patient-Related Factors on Speech Outcomes at
           5 Years of Age in Children With a Cleft Palate

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      Authors: Sophie Butterworth, Kate J Fitzsimons, Jibby Medina, Lorraine Britton, Stephanie Van Eeden, Hussain Wahedally, Min Hae Park, Jan van Der Muelen, Craig J H Russell
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo investigate the relationship between patient-related factors (sex, cleft type, cleft extent, and Robin Sequence [RS]) and speech outcome at 5 years of age for children born with a cleft palate ±  lip (CP ± L).Participants3157 Children (1426 female:1731 male) with a nonsyndromic CP ± L, born between 2006 and 2014 in England, Wales, and Northern Ireland.Outcome MeasurePerceptual speech analysis utilized the Cleft Audit Protocol for Speech–Augmented (CAPS-A) rating and UK National Speech Outcome Standards: Speech Standard 1 (SS1)—speech within the normal range, SS2a—no structurally related speech difficulties or history of speech surgery, and SS3—speech without significant cleft-related articulation difficulties.ResultsOdds of achieving SS1 were lower among boys (aOR 0.771 [CI 0.660-0.901]), those with clefts involving the lip and palate (vs palate only) (UCLP—aOR 0.719 [CI 0.591-0.875]; BCLP—aOR 0.360 [CI 0.279-0.463]), and clefts involving the hard palate (incomplete—aOR 0.701 [CI 0.540-0.909]; complete—aOR 0.393 [CI 0.308-0.501]). Similar relationships with these patient factors were observed for SS3. SS2 was affected by the extent of hard palate involvement (complete; aOR 0.449 [CI 0.348-0.580]). Although those with CP and RS were less likely to meet all 3 standards than those without RS, odds ratios were not significant when adjusting for sex and cleft extent.ConclusionSex, cleft type, and extent of hard palate involvement have a significant impact on speech outcome at 5 years of age. Incorporating these factors into risk-adjustment models for service-level outcome reporting is recommended.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-23T05:48:12Z
      DOI: 10.1177/10556656221110094
       
  • Does the Rigid External Distraction Device Alter Maxillary Pitch in Cleft
           Maxillary Distraction'

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      Authors: Alexander T. Mathews, Chad W. Dammling, Peter D. Waite, Brian E. Kinard
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe rigid external distraction (RED) device is reported to have the ability to three-dimensionally reposition the maxilla. The purpose of this study is to assess the ability of RED to intentionally alter the maxillary pitch.DesignRetrospective cohort study.SettingInstitutional practice.PatientsA retrospective chart review was completed over the past 12 years and a total of 50 patients met the inclusion criteria.Main outcome measuresCephalometric changes and alteration in palatal plane angle.MethodsCephalometric analysis of standardized landmarks was completed on calibrated, standardized lateral cephalograms. Pre-distraction and post-consolidation variables were compared via a two-tailed paired t-test.ResultsThe mean age at surgery of 12.2 ± 3.2 years. Through distraction osteogenesis (DO), the maxilla was moved anteriorly with a mean distraction distance of 8.4 ± 4.8 mm. The mean change in the angles sella-nasion-A-point angle (SNA), sella-nasion-B-point angle (SNB), and A-point-nasion-B-point angle (ANB) were 10.2 ± 4.8, 0.9 ± 2.7, and 9.3 ± 4.1, respectively. The mean change in the palatal plane angle was −4.4 ± 3.7. The mean change in the vertical position of the anterior nasal spine (ANS) and posterior nasal spine (PNS) in relation to the Frankfort horizontal (FH) were −2.0 ± 4.1 mm and 1.7 ± 3.8 mm, respectively.ConclusionThis study documents short-term findings of RED in a large cleft lip and palate (CLP) population. Despite positioning of distraction eyelets superior to the theoretical center of resistance, a counterclockwise (CCW) rotation of the palatal plane occurred. This suggests that adjunctive methods of vector control should be considered if clockwise (CW) rotation of the palatal plane is intended with the RED device.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-23T05:47:44Z
      DOI: 10.1177/10556656221109413
       
  • Presurgical Cleft Management of Infants: A Survey of ACPA Approved and
           International Cleft Palate and Craniofacial Teams

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      Authors: Sven W. C. Jensen, Emilija D. Jensen, Gabriella Kaminer-Levin, Camila Caro, Kyle Stevens
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo characterize the presurgical infant orthopedics (PSIO) and gingivoperiosteoplasty (GPP) protocols across the American Cleft Palate-Craniofacial Association (ACPA) approved and international cleft palate (CP) and craniofacial teams.DesignCross-sectional survey.SettingACPA approved and international CP and craniofacial teams.ResultsRespondents from 115 out of 215 ACPA approved and international CP and craniofacial teams permitted to contact (out of a total of 259 total teams) completed the survey (response rate  =  53.5%). There were 89 (77.4%) ACPA approved teams and the remaining international teams were mainly located in Europe (13.0%). Seventy-eight CP and craniofacial teams (67.8%) provided PSIO and 65 (83.3%) of these teams used alveolar molding (AM). Twenty-two CP and craniofacial teams (19.1%) provided GPP. A mean of 9.5  ±  2.6 different specialists were on the cleft team with the most common being orthodontists (97.4%), speech therapists (96.5%), and plastic/craniofacial surgeons (90.4%).ConclusionsMost ACPA approved and ACPA registered international CP and craniofacial teams provided PSIO techniques by orthodontists using lip taping (LT) and AM, while few provide GPP.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-21T05:11:47Z
      DOI: 10.1177/10556656221109416
       
  • Long-term Follow-up of Bilateral Cleft Lip and Palate: Incidence of
           Speech-Correcting Surgeries and Fistula Formation

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      Authors: Charlotta Gustafsson, Arja Heliövaara, Jorma Rautio, Junnu Leikola
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionWhile bilateral cleft lip and palate (BCLP) constitutes a clinical challenge for the whole cleft team, the ideal surgical protocol remains obscure. This study presents the long-term burden of care in terms of secondary surgeries, defined as fistula repair and speech-correcting surgeries (SCS), in a single center. Outcomes of two surgical protocols utilized over the years were also compared.Material and MethodsA retrospective single-center analysis of 81 non-syndromic children with complete BCLP born between 1990 and 2010. Two surgical protocols comprising single-stage and two-stage (delayed hard palate closure) procedures were compared. Outcome was analyzed at the time of alveolar bone grafting (ABG) and post-ABG.ResultsAltogether 54 children (66.7%) had underwent secondary surgery by the time of bilateral ABG. At this point, 38.3% (n = 31) of patients had received SCS and 49.4% (n = 40) had undergone fistula repair. The corresponding incidences at the end of follow-up were 46.9% (n = 38) and 53.1% (n = 43). No significant difference emerged in SCS incidence between the 2 protocols; however, prior to ABG the single-stage protocol had a significantly lower need for fistula repair. Regarding the location of fistulas, some differences were observed, with the single-stage procedure more associated with anterior fistulas.ConclusionBCLP has a high surgical burden of care in terms of secondary surgeries, defined as SCS and fistula repair. In our experience, the single-stage protocol, particularly the two-flap technique, offers better results in the management of BCLP than the two-stage approach with a short delay in hard palate closure.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-21T05:11:39Z
      DOI: 10.1177/10556656221102816
       
  • Longitudinal Morphological Changes in the Mucosal Free Margin After
           Unilateral Cleft Lip Repair

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      Authors: Yuta Nakajima, Shunsuke Yuzuriha, Fumio Nagai
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveWeakness of the mucosal free margin is common in secondary cleft lip deformities. However, the incidence is rarely reported after a long-term follow-up. We evaluated the evolution of patient mucosal free margin after primary repair and until the end of growth.DesignBlinded retrospective study (photography and chart reviews).SettingTertiary care hospital; private practice.PatientsForty-eight patients with unilateral cleft lip with or without cleft palate who underwent primary nasolabial repair between 1996 and 2004 were followed up until their end of growth marked at 18 and 16 years of age for male and female patients, respectively.InterventionsPrimary nasolabial repair using a straight-line closure without a triangular flap on the vermilion was performed; if needed, a revision surgery was performed at 5 years of age (85%).Main Outcome Measure:The contour of the mucosal free margin was classified into nodular, smooth, and recessed types at 1, 5, and 10 years and at the end of growth.ResultsAt 1 year of age, the nodular type was more common (58%). However, the recessed type was more common (75%) at the end of growth. The mucosal free margin continued to change after 10 years of age. At the end of growth, half of the patients with incomplete cleft experienced center weakness, and the others had cleft-side weaknesses.ConclusionsPatients with incomplete clefts showed favorable results at early ages. However, two-thirds of the patients with complete and incomplete clefts showed focal weakness at the end of growth.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-17T08:17:07Z
      DOI: 10.1177/10556656221108851
       
  • Global Trends in Knowledge, Attitude, and Awareness of Orthodontists
           Regarding the Management of Patients with Cleft lip and/or Palate: A
           Systematic Review

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      Authors: Sukeshana Srivastav, Nitesh Tewari, Shubhi Goel, Ritu Duggal, Gregory Antonarakis, Partha Haldar
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionThe aim of the systematic review is to assess the trends of knowledge, attitude, awareness, and practice related to these aspects among orthodontists globally.MethodsAn a priori protocol was developed as per the best practices of evidence-based medicine and registered in Prospero (CRD42022306107). The search was conducted electronically, using MeSH-terms, keywords, and Boolean-operators “AND” and “OR” in different combinations in multiple databases and Screening of titles and abstracts followed by the full-text evaluation was performed. The risk of bias (ROB) was assessed using Joanna Briggs Institute critical appraisal checklist.ResultsFive studies were included in the qualitative synthesis and three of them showed a high ROB. When participants enquired about which other specialists worked in the cleft team in addition to the orthodontists, 84% of them reported it to be general-dentist in one study. Furthermore, the absence of an interdisciplinary team was reported in two studies from Africa. When enquired about the percentage of their practice devoted to the care of cleft lip and palate (CLP)-patients, a study reported that 52% (n = 88) orthodontists had treated
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-17T05:58:04Z
      DOI: 10.1177/10556656221108856
       
  • Cranial Morphology Associated With Syndromic Craniosynostosis: A Potential
           Detection of Abnormality in Patient's Cranial Growth Using Angular
           Statistics

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      Authors: Nur Syahirah Zulkipli, Siti Zanariah Satari, Firdaus Hariri, Norli Anida Abdullah, Wan Nur Syahidah Wan Yusoff, Abdul Ghapor Hussin
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionApert, Crouzon, and Pfeiffer syndromes are common genetic syndromes related to syndromic craniosynostosis (SC), whereby it is a congenital defect that occurs when the cranial growth is distorted. Identifying cranial angles associated with these 3 syndromes may assist the surgical team to focus on a specific cranial part during the intervention planning, thus optimizing surgical outcomes and reducing potential morbidity.ObjectiveThe aim of this study is to identify the cranial angles, which are associated with Apert, Crouzon, and Pfeiffer syndromes.MethodsThe cranial computed tomography scan images of 17 patients with SC and 22 control groups aged 0 to 12 years who were treated in the University Malaya Medical Centre were obtained, while 12 angular measurements were attained using the Mimics software. The angular data were then divided into 2 groups (patients aged 0 to 24 months and >24 months). This work proposes a 95% confidence interval (CI) for angular mean to detect the abnormality in patient's cranial growth for the SC syndromes.ResultsThe 95% CI of angular mean for the control group was calculated and used as an indicator to confirm the abnormality in patient's cranial growth that is associated with the 3 syndromes. The results showed that there are different cranial angles associated with these 3 syndromes.ConclusionsAll cranial angles of the patients with these syndromes lie outside the 95% CI of angular mean of control group, indicating the reliability of the proposed CI in the identification of abnormality in the patient's cranial growth.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-17T05:57:24Z
      DOI: 10.1177/10556656221107524
       
  • Impact of Illustrated Postoperative Instructions on Knowledge and
           Retention During a Cleft Lip and Palate Surgical Mission

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      Authors: Elizabeth B. Card, Carrie E. Morales, Juan M. Ramirez, Marce Billingslea, Ariel Marroquín, Eo Trueblood, Luv R. Javia, Susan M. McCormack, Leonard R. Friedland, David W. Low, Alan Jay Schwartz, Michelle Scott, Oksana A. Jackson
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo determine the impact of illustrated postoperative instructions on patient-caregiver knowledge and retention.DesignProspective study with all participants receiving an educational intervention.SettingPediatric plastic surgical missions in Guatemala City, Guatemala, between 2019 and 2020.ParticipantsA total of 63 majority-indigenous Guatemalan caregivers of patients receiving cleft lip and/or palate surgery.InterventionIllustrated culturally appropriate postoperative care instructions were iteratively developed and given to caregivers who were surveyed on illustration-based and text-based information at preoperative, postoperative, and four-week follow-up time points.Main Outcome MeasurePostoperative care knowledge of illustration-based versus text-based information as determined by the ability to answer 11 illustration- and 8 text-based all-or-nothing questions, as well as retention of knowledge as determined by the same survey given at four weeks follow-up.ResultsScores for illustration-based and text-based information both significantly increased after caregivers received the postoperative instructions (+13.30 ± 3.78 % SE, + 11.26 ± 4.81 % SE; P  .05), but significantly lower for text-based information (−28.46 ± 6.09 % SE, P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-17T05:57:15Z
      DOI: 10.1177/10556656221100052
       
  • BMP4 Regulates EMT to be Involved in non-Syndromic Cleft lip With or
           Without Palate

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      Authors: Jia-Wei Hong, Yue Yu, Lu-Shan Wang, Zheng Li, Rui Zhang, Qi Wang, Zhen Ding, Jin-Peng Zhang, Mei-Rong Zhang, Li-Chun Xu
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveIn the previous study, we identified bone morphogenetic protein 4 (BMP4) responsible for non-syndromic cleft lip with or without cleft palate (NSCL/P). We aimed to elucidate the effects and mechanisms of BMP4 on epithelial–mesenchymal transition (EMT) through Smad1 signaling pathway to be involved in NSCL/P.MethodsThe human oral epidermoid carcinoma cells (KBs) were transfected with plasmids or small interfering RNA (siRNA) to build the models. The migration of the cells was evaluated by transwell assay. Western blotting and quantitative real-time reverse transcription-polymerase chain reaction (qRT-PCR) were used to detect the expressions of BMP4, E-cadherin, N-cadherin, EMT-related transcription factors snal1 and snal2, matrix metalloproteinase 2 (MMP2), MMP9, Smad1, and phosphorylated Smad1.ResultsIn the overexpression group, the migration number of cells was increased significantly. The protein expression of E-cadherin was decreased significantly, while the protein expression level of the N-cadherin was increased significantly. The protein and mRNA expressions of MMP2, MMP9, snal1, and snal2 were significantly higher. The expression level of Smad1 was not significantly changed, while the phosphorylation of Smad1 was significantly increased. In the BMP4-siRNA group, the migrating number cells was significantly decreased. The protein expression of E-cadherin was increased significantly, while the expression of N-cadherin was significantly decreased. The protein and mRNA expressions of MMP2, MMP9, snal1, and snal2 were significantly lower than that of the control group. The expressions of Smad1 and phosphorylation of Smad1 were not significantly changed.ConclusionsBMP4 enhances cell migration and promotes cell EMT through Smad1 signaling pathway. Abnormal BMP4 mediates migration and EMT through other relevant signaling pathways resulting in NSCL/P. The study provides new insight into the mechanisms of NSCL/P associated with BMP4.n
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-15T05:25:55Z
      DOI: 10.1177/10556656221105762
       
  • Pleomorphic Adenoma of the Palate: A Rare Case in an Adolescent With Cleft
           lip and Palate

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      Authors: David Sterling, Alison Kaye, Michael Lypka
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Benign salivary gland tumors are rarely found in children and adolescents compared with adults. Pleomorphic adenomas (PAs), the most common benign salivary gland tumor, account for only 1% of all head and neck lesions and fewer than 5% of all salivary gland tumors in individuals under the age of 16 years. The data on palatal PA in the first 2 decades of life is confined to published case reports and case series. To date, there has never been a report of palatal PA in a patient with cleft lip and palate. Here we describe an adolescent female with bilateral cleft lip and palate with PA of the hard and soft palate who underwent wide local excision and reconstruction with a buccal fat pad and buccal myo-mucosal flap.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-15T05:25:45Z
      DOI: 10.1177/10556656221102038
       
  • Ten-Year Cephalometric Comparison of Patients With Cleft Palate who
           

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      Authors: Katie Garland, Michelle Coyle, Tim Foley, Damir Matic
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundPre-surgical orthopedic (PSO) devices can be used in the management of patient with cleft lip/palate (CL/P) to narrow the alveolar gap (AG) prior to lip surgery. There are few studies comparing these 2 devices. The objective of this work was to compare the effects of active and passive PSO devices on facial growth in a single surgeon's cohort of patients with CL/P over a 10-year period.MethodsA retrospective review of all patients with CL/P in a single surgeon's practice from 2002 to 2018 was performed. Preoperative measurements of AG size were done using electronic calipers on patient molds. Patient radiographs were taken at 5 and 10 years of age and cephalometric landmarks were plotted using specialized software. Independent sample t-tests were used to compare means for maxillary, mandibular, vertical, and dento-alveolar growth parameters.ResultsTwenty patients with an active device and 23 patients with a passive device were included. No differences were observed in the basic demographic information between the two groups. At the time of lip repair, patients with a passive device had significantly larger horizontal AGs (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-14T06:00:23Z
      DOI: 10.1177/10556656221106891
       
  • VPI Management in SATB2 Syndrome: Use of MRI to Evaluate Anatomy and
           Physiology in Non-Cleft VPI

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      Authors: Jamie L. Perry, Jessica L. Williams, Taylor D. Snodgrass, Thomas J. Sitzman
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      This clinical case study describes the velopharyngeal anatomy and physiology in a patient who presented with SATB2-associated syndrome (SAS) and velopharyngeal insufficiency (VPI) in the absence of an overt cleft palate. The clinical presentation, treatment, outcome, and the contribution of anatomical findings from MRI to surgical treatment planning for this rare genetic disorder, SAS, are described. This case study contributes to our current understanding of the anatomy and physiology of the velopharyngeal mechanism in an individual born with SAS and non-cleft VPI. It also details the changes following bilateral buccal myomucosal flaps in this patient.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-13T06:19:44Z
      DOI: 10.1177/10556656221106888
       
  • Speech Telepractice and Treatment Intensity in a Cantonese-Speaking Case
           with 22q11.2 Deletion Syndrome Following Late Diagnosis and Management of
           Velopharyngeal Dysfunction

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      Authors: Valerie J. Pereira, Janet Y.T. So, Joy M.K. Tsang, Wing S. Choi, Michael C.F. Tong, Kathy Y.S. Lee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      This case report explores clinical treatment efficacy in a Cantonese-speaking child with 22q11.2 Deletion Syndrome where diagnosis and management of velopharyngeal dysfunction can be considered late. All treatment sessions were undertaken via telepractice during the peak of the COVID-19 pandemic in Hong Kong. A hybrid of specialized cleft palate speech treatment techniques and traditional treatment approaches in Speech Sound Disorders were utilized. Treatment intensity components including dose, dose form, session duration, and total intervention duration were documented.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-09T01:13:48Z
      DOI: 10.1177/10556656221106042
       
  • Bone Tissue Engineering Strategies for Alveolar Cleft: Review of
           Preclinical Results and Guidelines for Future Studies

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      Authors: Jenn J. Park, Danielle H. Rochlin, Yassmin Parsaei, Pradip R. Shetye, Lukasz Witek, Philipp Leucht, Piul S. Rabbani, Roberto L. Flores
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      The current standard of care for an alveolar cleft defect is an autogenous bone graft, typically from the iliac crest. Given the limitations of alveolar bone graft surgery, such as limited supply, donor site morbidity, graft failure, and need for secondary surgery, there has been growing interest in regenerative medicine strategies to supplement and replace traditional alveolar bone grafts. Though there have been preliminary clinical studies investigating bone tissue engineering methods in human subjects, lack of consistent results as well as limitations in study design make it difficult to determine the efficacy of these interventions. As the field of bone tissue engineering is rapidly advancing, reconstructive surgeons should be aware of the preclinical studies informing these regenerative strategies. We review preclinical studies investigating bone tissue engineering strategies in large animal maxillary or mandibular defects and provide an overview of scaffolds, stem cells, and osteogenic agents applicable to tissue engineering of the alveolar cleft. An electronic search conducted in the PubMed database up to December 2021 resulted in 35 studies for inclusion in our review. Most studies showed increased bone growth with a tissue engineering construct compared to negative control. However, heterogeneity in the length of follow up, method of bone growth analysis, and inconsistent use of positive control groups make comparisons across studies difficult. Future studies should incorporate a pediatric study model specific to alveolar cleft with long-term follow up to fully characterize volumetric defect filling, cellular ingrowth, bone strength, tooth movement, and implant support.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-09T01:13:40Z
      DOI: 10.1177/10556656221104954
       
  • Comparison of Maxillofacial Morphology Between Modified Furlow's and
           Modified two-Flap Palatoplasty in Orofacial Clefts During the Primary
           Dentition Period

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      Authors: Hiroshi Yoshida, Masahiro Takahashi, Tetsutaro Yamaguchi, Hideomi Takizawa, Momoko Takakaze, Koutaro Maki
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo assess the effect of two palatoplasty procedures, modified Furlow's palatoplasty (F procedure) and modified two-flap palatoplasty (T procedure), on the maxillofacial morphology of unilateral cleft lip and palate (UCLP) and bilateral cleft lip and palate (BCLP) during the primary dentition period.DesignRetrospective cohort studySettingDepartment of Orthodontics, School of Dentistry, Showa UniversityParticipantsWe enrolled 106 pediatric patients (63 boys, 43 girls; aged 4.43  ±  0.34 years) with non-syndromic orofacial clefts who underwent cheiloplasty and palatoplasty.InterventionsPatients were divided into four groups according to cleft type (BCLP or UCLP) and palatoplasty procedure type (F or T procedure).Main Outcome MeasuresMaxillofacial morphology was assessed by examining parameters on lateral cephalograms.ResultsMultiple comparisons revealed significant differences among N-A, N-ANS, and ANS-PNS distances and SNA and ANB angles among the groups. There were significant differences in N-A, N-ANS, ANS-PNS, SNA, and SNB among the UCLP and BCLP groups. The calculated effect sizes were all within 0.3–0.5. The statistical power was as follows: N-A, 86.41%; N-ANS, 79.77%; ANS-PNS, 97.49%; SNA, 96.88%; and ANB, 99.25%.ConclusionsAlthough UCLP and BCLP both exhibited differences in craniofacial distances and angles, the procedure type (either F or T procedure) had no significant effect on the maxillofacial morphology (as determined by lateral cephalograms). However, to rule out additional bias, patient-specific factors should be considered that may be affected by maxillofacial development when deciding surgical approaches.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-08T05:50:43Z
      DOI: 10.1177/10556656221104374
       
  • A Multisite Study Investigating Child and Parent Proxy Reported Quality of
           Life in Children With Cleft Lip and/or Palate

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      Authors: Celia E. Heppner, Canice E. Crerand, Claudia Crilly Bellucci, Farah Sheikh, Suzanne Woodard, Meredith Albert, Amy L. Conrad, Kathleen A. Kapp-Simon
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis observational, multisite cohort study explored health-related quality of life (HRQoL) in children with cleft lip and/or palate (CL/P), including interrater agreement and ratings for this group relative to clinical cutoff scores and published means for healthy and chronically ill children.MethodsParticipants (338 children ages 8-10 years, 45.9% male and their parents, 82.0% female) across 6 sites completed the Pediatric Quality of Life Inventory Generic Core Scales (PedsQL).ResultsIntraclass correlation revealed poor interrater agreement for most HRQoL domains. Although ratings were generally higher than those expected for children with a chronic illness, child ratings were below healthy means for school functioning, and parent proxy ratings were below healthy means for all domains except physical functioning. Lower ratings consistent with chronic illness means were found for self-reported emotional and psychosocial functioning in children with cleft lip and palate (CLP), as well as parent proxy-reported emotional, school, and psychosocial functioning for children with cleft palate (CP). Scores were most likely to be in the clinical range for children with CP for social, school, and total functioning.ConclusionAlthough parent proxy report provides important information about observed functioning, poor interrater agreement indicates that both child and parent proxy reported HRQoL should be included in outcomes assessment for CL/P. HRQoL ratings may be higher for children with CL/P compared to youth with other chronic illnesses, but psychosocial functioning may be negatively impacted when compared with healthy youth, particularly for emotional, social, and school functioning in children with CLP or CP.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-08T04:29:17Z
      DOI: 10.1177/10556656221105766
       
  • Effects of RME on Hearing in UCLP Patients: A Pilot Study

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      Authors: Kaleem Fatima, Prabhat Kumar Chaudhari, Ritu Duggal, Om P. Kharbanda, Alok Thakar
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo assess the changes in hearing after rapid maxillary expansion (RME) and at the end of 6 months retention period in complete unilateral cleft lip and palate (UCLP) patients using pure tone audiometry and tympanometry test.DesignProspective pilot study.SettingTertiary health care teaching hospital in New Delhi.PatientsThis study was conducted on 6 UCLP patients in the age range of 6-14 years with normal ear anatomy. All 6 patients had undergone primary repair of cleft lip and palate and required no more than 5 mm expansion in the intermolar region.InterventionRME was done using a Hyrax expander with daily activation of one-quarter turn per day for a period of 15-20 days (0.25 mm per day).Main outcome measureAudiometry and tympanometry readings at the baseline as compared to the post expansion and at the end of 6 months retention period.ResultsThere were no significant changes in the hearing levels on the audiometry test after RME on the cleft side (p-value −0.51) and the noncleft side ear (p-value −0.26). No significant changes were observed in the middle ear volume on the tympanometry test after RME on the cleft side (p-value −0.09) and the noncleft side ear (p-value −0.28).ConclusionThere was no improvement or deterioration in the hearing levels after RME and at the end of 6 months retention period in UCLP patients as evaluated using pure tone audiometry and tympanometry test. Hence RME may be undertaken in UCLP patients safely in terms of hearing is concerned.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-08T04:28:57Z
      DOI: 10.1177/10556656221104945
       
  • Total Aplasia of Paranasal Sinus Associated With a Syndromic Condition

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      Authors: Emrah Doğan, Erdoğan Özgür
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Total aplasia of paranasal sinus (TAPS) is extremely rare, although the fact that partial aplasia is very common. TAPS seems to be limited to only 5 case reports in the literature until now. We present the case of a 29-year-old patient who has a syndromic face appearance but whose TAPS was detected incidentally. The maxillary, sphenoid, ethmoid, and frontal sinuses were totally aplastic. Furthermore, clinodactyly and high-arched palate were observed. The patient's appearance was consistent with a syndromic face because of some findings on inspection such as hypertelorism, shortening of the palpebral fissure, protruded and wide nasal base, high arched palate and zygomatic hypoplasia. The patient's profile was more suitable for Teacher-Collins syndrome than other syndromes, however, a certain diagnosis was not made genetically. To the best of our knowledge, this is the first reported association between TAPS and a syndromic condition.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-08T04:28:48Z
      DOI: 10.1177/10556656221074865
       
  • Prevalence and Management of Laryngomalacia in Patients With Pierre Robin
           Sequence

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      Authors: Anna E. Bakeman, Amber D. Shaffer, Allison B. J. Tobey, Noel Jabbour, Matthew D. Ford, Jesse A. Goldstein, Jeffrey P. Simons
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo characterize the prevalence and presentation of laryngomalacia and efficacy of supraglottoplasty (SGP) in a cohort of patients with Pierre Robin Sequence (PRS).DesignRetrospective cohort study.SettingTertiary-care children's hospital.Patients, ParticipantsConsecutive patients with PRS born between January 2010 and June 2018.Main Outcome MeasuresChart review included demographics, comorbid airway obstruction including laryngomalacia, timing of surgical interventions, clinical symptoms, sleep study data, and modified barium swallow study data.Results126 patients with PRS were included; 54% had an associated syndrome, 64% had an overt cleft palate, and 22% had a submucous cleft palate. 64/126 were noted to have laryngomalacia (51%). Patients with concurrent PRS and laryngomalacia were significantly more likely to have submucous cleft palate (P = .005) and present with aspiration with cough (P = .01) compared to patients with PRS without laryngomalacia. Patients with concurrent laryngomalacia and PRS showed a significant decrease in apnea–hypopnea index (AHI) and obstructive AHI (OAHI) after mandibular distraction, with a median AHI and OAHI improvement of 22.3 (P = .001) and 19.8 (P = .002), respectively. Patients who underwent only SGP did not show significant improvement in these parameters (P = .112 for AHI, P = .064 for OAHI).ConclusionsThe prevalence of laryngomalacia in our PRS cohort was 51%. Patients with PRS and laryngomalacia are more likely to present with overt aspiration compared to patients with PRS without laryngomalacia. These data support that laryngomalacia does not appear to be a contraindication to pursuing MDO.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-07T05:17:42Z
      DOI: 10.1177/10556656221107298
       
  • Trends in Cleft lip and/or Palate Prevalence at Birth in Mexico: A
           National (Ecological) Study Between 2003 and 2019

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      Authors: Eduardo Cerón-Zamora, Rogelio José Scougall-Vilchis, Rosalía Contreras-Bulnes, Blanca Silvia González-López, Miriam Alejandra Veras-Hernández, Salvador Eduardo Lucas-Rincón, Mauricio Escoffié-Ramirez, Carlo Eduardo Medina-Solís, Gerardo Maupomé
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo describe trends in cases and prevalence at birth rates of cleft lip and/or palate (CL/P) in Mexico between 2003 and 2019.DesignEcological study.SettingMultiple data sources systematically collected into a national epidemiological surveillance data warehouse.ParticipantsNational Live Birth Information System.Main Outcome Measure(s)Both cases and prevalence at birth rates of CL/P in Mexico within a 17-year period were used as dependent variables.ResultsAt the national level there were 23 184 new cases of CL/P (average of 1364 per year) in the 32 states of Mexico, with an average prevalence at birth rate of 0.53 per 1000 live births. The states with the highest prevalence at birth rates of CL/P during the period were Hidalgo (1.59) and Jalisco (1.32), while the states with the lowest rates were Nayarit (0.22) and Durango (0.29). A slight decrease in both cases (z = −2.41, P = .016) and prevalence at birth rates (z = −2.58, P = .010) of CL/P was observed at the national level. States such as Durango, Puebla, Chiapas, Guerrero, Oaxaca, Mexico City, State of Mexico, Coahuila and Jalisco showed a clear downward trend (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-07T05:17:21Z
      DOI: 10.1177/10556656221106881
       
  • Comparison of Cleft Lip Nasal Deformities Between Lesser-Form and
           Incomplete Cleft Lips: Implication for Primary Rhinoplasty

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      Authors: Young Chul Kim, Dae Won Hong, Tae-Suk Oh
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study investigated various manifestations of nasal deformities in lesser-form cleft lips, including the minor-form, microform, and mini-microform, by photogrammetric comparison with incomplete cleft lips.DesignRetrospective study.SettingTertiary university-affiliated hospital.ParticipantsA total of 160 patients with unrepaired unilateral incomplete cleft lips ranging from lesser-form to two-thirds way clefts.Main Outcome MeasuresThe severity of nasal deformities was assessed by photogrammetric measurements of linear and angular variables. The symmetry ratio between the cleft and non-cleft sides was obtained by measuring various nasal parameters and comparing them among the different labial cleft groups.ResultsThe degree of nasal deformities increased with the extent of labial clefts among the 3 labial cleft groups (lesser-form, halfway, and two-thirds way clefts) in terms of alar base width ratio (1.102, 1.197, 1.309; P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-01T05:55:19Z
      DOI: 10.1177/10556656221105204
       
  • Microbial Analysis of Obturators During Maxillofacial Prosthodontic
           Treatment Over an 8-Year Period

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      Authors: Karl M Lyons, Richard D Cannon, John Beumer, Mahmoud M Bakr, Robert M Love
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe aim of the study was to investigate the microbial colonization (by Candida species, anaerobic and facultative anaerobic bacteria) of maxillary obturators used for the restoration of maxillary defects, including during radiotherapy.Design:Retrospective cohort study.ParticipantsFifteen patients requiring a maxillary obturator prosthesis had swabs of their obturators and adjacent tissues taken at different stages of their treatment over a period of 8 years.MethodIdentification of microbial species from the swabs was carried out using randomly amplified polymorphic DNA polymerase chain reaction (RAPD PCR) analysis, checkerboard DNA–DNA hybridization, CHROMagar Candida chromogenic agar, and DNA sequencing.ResultsCandida species were detected in all patients and all patients developed mucositis and candidiasis during radiotherapy which was associated with an increase in colonization of surfaces with Candida spp., particularly C albicans. Microbial colonization increased during radiotherapy and as an obturator aged, and decreased following a reline, delivery of a new prosthesis, or antifungal treatment during radiotherapy.ConclusionsMicrobial colonization of maxillary obturators was related to the stage of treatment, age of the obturator material, radiotherapy and antifungal medications, and antifungal treatment may be recommended if C albicans colonization of palatal tissues is greater than 105 colony-forming units per cm2 following the first week of radiotherapy.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-01T05:54:58Z
      DOI: 10.1177/10556656221104940
       
  • Differences in Practice in Alveolar Bone Grafting Among American Cleft
           Palate-Craniofacial Association Members

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      Authors: Eric Zielinski, Chiara N. Santiago, Gaia S. Santiago, Ian Zelko, Robert Hlavin, Akriti Choudhary, Chad A. Purnell
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe purpose of this study is to determine areas of agreement and disagreement among American Cleft Palate-Craniofacial Association (ACPA)members in the clinical practice of alveolar bone grafting (ABG), to guide further research to optimize ABG practices.DesignA cross-sectional survey was conducted.SettingThe respondents were in an academic, combination, or private practice.Patients, participantsThe respondents were either plastic or oral and maxillofacial surgeons (OMFS) from various countries.InterventionsA de-identified 24-question online survey was distributed to ACPA surgeon members utilizing the Research Electronic Data Capture (REDCap) tool.Main outcome measureData collected included surgeon specialty, use of various alveolar bone graft surgical techniques, as well as protocols.ResultsThere was more variability than consensus between specialties with regards to the preoperative workup, timing of surgery, materials used for bone graft, surgical techniques, perioperative management, and postoperative evaluation. There was consensus on grafting during mixed dentition, not staging soft and hard tissue closure, and using iliac crest for primary and secondary grafting. Disagreements involved factors used to time the procedure and type of imaging used to assess viability. Technical differences involved incision type, part of bone grafted, use of minimally invasive technique, and material used for revisions.ConclusionsAside from areas of consensus among surgeons on ABG, several areas, including use of bone substitutes in revision grafting, incision and type of iliac crest graft used during initial grafting, and postoperative protocols, had no consensus. These areas should be targets of further research to determine if there truly is an optimal method to perform ABG.MethodsThe study was approved by the University of Illinois at Chicago Institutional Review Board. A de-identified 24-question online survey was distributed to surgeon members of the ACPA utilizing the REDCap tool on August 7, 2020. The survey questions consisted of multiple choice and multiple selection questions including an option to select “other” and specify the information in a blank space. Data collected included surgeon specialty, use of various alveolar bone graft surgical techniques, as well as pre and postoperative protocols. The full survey is included in online Supplemental material. Data analysis was performed in SPSS Statistics 27 (IBM Corp.). Descriptive statistics were performed, and chi-square was used to test for significant differences in survey responses between groups.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-01T05:54:38Z
      DOI: 10.1177/10556656221104036
       
  • Impact of Syndromes on Sleep-Disordered Breathing in Children After Cleft
           Palate Repair

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      Authors: Nicolas S. Poupore, W. Nicholas Jungbauer, Hussein Smaily, William W. Carroll, Phayvanh P. Pecha
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivePrior research suggests that children with cleft palate (CP) are at increased risk of obstructive sleep-disordered breathing (SDB). However, few studies differentiate the effects of CP repair on SDB based on syndrome status. The goal of this study was to evaluate differences in SDB after palatoplasty among children with nonsyndromic CP, syndromic CP, and isolated Robin sequence (RS).DesignRetrospective chart review.SettingTertiary academic children's hospital.Patients/ParticipantsA total of 145 children who underwent primary CP repair from 2014 to 2021.Main Outcome MeasurePost-palatoplasty SDB is defined as parent-reported symptoms and/or evidence of obstructive sleep apnea (OSA).ResultsMedian age at palatoplasty was 11.1 [IQR 10.2-13.6] months. Most patients (61.4%) had nonsyndromic CP, 26.9% had a syndrome, and 11.7% had RS. Children with syndromic CP and RS had more post-palatoplasty SDB symptoms (56.4% vs 58.8% vs 30.3%, P = .006) and higher rates of OSA (25.6% vs 29.4% vs 5.6%, P = .001) compared to children with nonsyndromic CP after palatoplasty. Children with syndromic CP and RS had nearly 3 to 4 higher odds of post-palatoplasty SDB than children with nonsyndromic CP (adjusted odds ratio [aOR] 2.88, 95% CI 1.29–6.47, P = .010; aOR 3.73, 95% CI 1.19–11.70, P = .024).ConclusionThis study showed that children with CP experience higher rates of SDB after palatoplasty than the general pediatric population. Within the cohort, children with syndromic CP and isolated RS were more likely to have obstructive sleep disorders than nonsyndromic children after palatoplasty. Clinicians should counsel caregivers accordingly and closely monitor these groups for SDB after palate repair.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-01T05:36:33Z
      DOI: 10.1177/10556656221105203
       
  • American Indian and Alaska Native Accessibility to Comprehensive Cleft Lip
           and Palate Treatment

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      Authors: Erik M. Wolfswinkel, Anna C. Howell, Beau MacDonald, John P. Wilson, Lori K. Howell
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveAmerican Indians and Alaska Natives (AI/AN) have the highest incidence of cleft lip and palate (orofacial clefts [OFCs]) when compared to other ethnic groups. We aim to determine the AI/AN populations’ proximity and accessibility to American Cleft Palate-Craniofacial Association accredited centers (ACPA centers) for treatment of OFCs. Our hypothesis is an unacceptable proportion of the AI/AN population lacks reasonable accessibility to ACPA centers and comprehensive craniofacial care.DesignA cross-sectional study of ACPA centers and AI/AN populations were analyzed for possible disparities.Main Outcome MeasurementsACPA centers were mapped using Geographic Information Systems (GIS) and compared with 2018 census population data and 2017 Tribal Census Tract data to visually display possible disparities. Total annual potential pediatric cleft care need for selected high-density AI/AN populated lands were estimated.ResultsGIS mapping demonstrates geographical isolation of AI/AN populations from ACPA centers. Two states with high AI/AN populated lands (ND, WY) have no ACPA centers. 47.1% of ACPA centers in high AI/AN populated lands have no craniofacial trained surgeons versus 78.9% craniofacial staffed ACPA centers nationally. The potential unmet cleft and craniofacial care need in selected high-density AI/AN populated lands is 1042 children.ConclusionAI/AN populations are likely underserved by ACPA centers and by craniofacial fellowship-trained staffed centers. Not addressing OFCs with comprehensive care can lead to worsened outcomes and further marginalization of these children. With future studies, we will be capable of making data-driven, informed decisions to more effectively ensure AI/AN access to comprehensive cleft and craniofacial care.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-01T05:36:19Z
      DOI: 10.1177/10556656221104942
       
  • Perioperative Pain Management After Primary Palate Repair: A 3-Surgeon
           Retrospective Study

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      Authors: Ingrid M. Ganske, Olivia C. Langa, Alex T. Cappitelli, Laura C. Nuzzi, Steven J. Staffa, Nancy DiTullio, Zoe Fullerton, Walid Alrayashi, John G. Meara, Carolyn R. Rogers-Vizena
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivePain management strategies following palatoplasty vary substantially. Despite efforts to reduce narcotic utilization, specific analgesic regimens are typically guided by surgeon preference. Our aim was to define analgesic variables that affect postoperative narcotic use and time to resumption of oral intake.DesignThis is a retrospective review from 2015 to 2018.PatientsNonsyndromic patients undergoing primary palate repair.Main Outcomes MeasuresAnalgesic variables included: local anesthetic, pterygopalatine ganglion nerve block, palatal pack, and postoperative use of ketorolac, dexamethasone, and nursing-controlled analgesia (NCA) opioid dosing. Proxy measures for pain included time to resumption of oral intake and morphine equivalence (mg/kg/h) administered.ResultsVeau phenotypes for the 111 patients included were: I (28%), II (19%), III (33%), IV (16%), and submucous (4%). Age, weight, local anesthetic, and postoperative use of ketorolac, dexamethasone, and palatal pack had no effect on either proxy measure (P > .05). Postoperative narcotic usage was significantly lower in patients who had an intraoperative suprazygomatic peripheral nerve block and significantly higher when NCA was utilized (P  .05).ConclusionSeveral perioperative analgesic strategies lead to comparable postoperative consumption of narcotic and time to resume oral intake. The authors advise careful consideration of NCA due to the potential for increased narcotic utilization that we found in our institution. Based on our promising findings, further studies are warranted to assess risks, benefits, and costs of performing peripheral nerve blocks at the time of palatoplasty.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-06-01T03:31:31Z
      DOI: 10.1177/10556656221075932
       
  • A Survey on Enhanced Recovery After Surgery (ERAS) Elements in Cleft
           Palate Repair

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      Authors: Christina Grabar, Jennifer Fligor, Melissa Kanack, Juleah Walsh, Joe Kim, Raj Vyas
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aims to characterize current use, knowledge, and attitude toward ERAS protocols by academic craniofacial surgeons.DesignCraniofacial surgeons were provided with electronic surveys.SettingElectronic survey; Institutional tertiary surgeons.Participants102 cleft palate surgeons surveyed and 31 completed the survey (30.4%).InterventionsNone.Main Outcome MeasuresRespondents rated their knowledge, use, and willingness to implement perioperative interventions modeled after adult ERAS protocols.ResultsMajority (67.7%) rated they were knowledgeable about ERAS. However, 61.3% “never use” a standardized protocol for cleft palate surgery. Only 3 ERAS elements are currently implemented by a majority of cleft surgeons: avoiding prolonged perioperative fasting (67.7%), using hypothermia prevention measures (74.2%), and minimizing use of opioids (62.5%). A large majority of respondents noted they never administer bolus (71.0%) or infusion (80.6%) dosing of tranexamic acid; most of these surgeons also indicated that administering tranexamic acid “would not be a valuable addition” (67.7% and 71.0%, respectively). Short-acting sedatives are used by 12.9% and by 16.1% of surgeons in all patients during extubation and postoperative recovery, respectively. By contrast, 22.6% never use such agents during extubation and 48.4% never use it during postoperative recovery. Overall, 67.7% of respondents replied that they would be willing to implement an ERAS protocol for cleft palate repair.ConclusionsMany respondents report using interventions compatible with an ERAS approach and the majority are willing to implement an ERAS protocol for cleft palate repair.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-27T05:31:42Z
      DOI: 10.1177/10556656221103756
       
  • Gender Authorship Trends Among Craniofacial Publications: A 20-Year
           Analysis

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      Authors: Fei Wang, Tiffany Cheng, Joseph A. Ricci
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aims to identify gender disparities within the subspecialty of craniofacial surgery as women surgeons remain underrepresented in academia and leadership, arenas heavily dictated by research productivity.DesignAll craniofacial articles published in 3 major research journals from 2000 to 2020 were reviewed and evaluated in 5-year increments.OutcomesInformation regarding author gender, authorship distribution, geographic origin, and publication type was collected. ANOVA, χ2, and logistic regression modeling were used for analysis.ResultsIn total, there were 3684 articles with 15 206 total authors—3128 (20.6%) were women, including 665 (21.3%) first authors, 1980 (63.2%) middle authors, and 487 (15.7%) senior authors. Mean women authorship increased significantly from 2000 to 2020 (0.33 vs 1.22 P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-25T03:29:50Z
      DOI: 10.1177/10556656221102040
       
  • Squamosal Suture Synostosis: An Under-Recognized Phenomenon

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      Authors: Justin W. Beiriger, Xiao Zhu, Madeleine K. Bruce, Zhazira Irgebay, John Smetona, Joseph E. Losee, Jesse A. Goldstein
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionThe squamosal suture (SQS) joins the temporal to the parietal bones bilaterally and is a poorly described site of craniosynostosis. SQS fusion is thought to occur as late as the fourth decade of life and beyond; however, we have incidentally noted its presence among our pediatric patients and hypothesize that it may occur earlier in life and more frequently than previously believed.MethodsA retrospective review of imaging performed on pediatric patients was completed to identify patients with SQS synostosis. This included a review of clinical notes as well as computed tomography (CT) images obtained by our craniofacial clinic. Relevant patient data and imaging were reviewed.ResultsForty-seven patients were identified with SQS synostosis, 21 were female (45%). Age at the time of radiographic diagnosis was 10.1 ± 8.4 years (range 17 days to 27 years). A majority of patients had bilateral SQS synostosis (57%), with a relatively even distribution of unilateral right (23%) versus left (19%). SQS was an isolated finding (no other suture involvement) in 15 patients (32%), all of whom were normocephalic and did not require surgical intervention. Thirty-two patients (68%) had concomitant craniosynostosis of other sutures, most commonly sagittal and coronal. Nine patients (19%) underwent surgery to correct cranial malformations—all these patients had multi-suture synostosis (P = 0.012). Twenty-seven patients (57%) had SQS synostosis diagnosed incidentally compared to 20 (43%) who were imaged with suspicion for synostosis. In those who were symptomatic, common findings included developmental delay, elevated intracranial pressure, hydrocephalus, seizures, and visual/hearing impairments. Ten patients (21%) were syndromic, the most frequent of which was Crouzon syndrome. No single pattern of calvarial malformation could be definitively described for SQS synostosis.ConclusionGiven that most isolated SQS synostosis cases were normocephalic, asymptomatic, and discovered incidentally, it is likely that there are many cases of unidentified SQS synostosis. The significance of SQS synostosis is currently unclear, and warrants further investigation into this phenomenon, its natural course, and its potential presence in the spectrum of normal development.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-20T08:13:31Z
      DOI: 10.1177/10556656221100675
       
  • Closing the Gap: A Systematic Review and Meta-Analysis of Enhanced
           Recovery After Surgery Protocols in Primary Cleft Palate Repair

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      Authors: Paul A. Asadourian, Marcos Lu Wang, Michelle R. Demetres, Thomas A. Imahiyerobo, David M. Otterburn
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveAssess the evidence for Enhanced Recovery After Surgery (ERAS) protocols in the cleft palate population.DesignA systematic review of MEDLINE, Embase, Cochrane, and CINAHL databases for articles detailing the use of ERAS protocols in patients undergoing primary palatoplasty.SettingNew York-Presbyterian Hospital.Patients/ParticipantsPatients with cleft palate undergoing primary palatoplasty.InterventionsMeta-analysis of reported patient outcomes in ERAS and control cohorts.Main Outcome Measure(s)Methodological quality of included studies, opioid use, postoperative length of stay (LOS), rate of return to emergency department (ED)/readmission, and postoperative complications.ResultsFollowing screening, 6 original articles were included; all were of Modified Downs & Black (MD&B) good or fair quality. A total of 354 and 366 were in ERAS and control cohorts, respectively. Meta-analysis of comparable ERAS studies showed a difference in LOS of 0.78 days for ERAS cohorts when compared to controls (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-18T07:24:37Z
      DOI: 10.1177/10556656221096631
       
  • LeFort III/I for Beckwith–Wiedemann Syndrome: A Case Report

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      Authors: John N. Muller, Pradip R. Shetye, Roberto L. Flores
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      This case presents a facially mature patient with Beckwith–Wiedemann Syndrome (BWS) who presented with severe class III malocclusion. Computed tomography imaging revealed an anterior crossbite of 19 mm and a narrow pharyngeal airway at the level of the tongue base precluding mandibular setback surgery. The patient was indicated for a LeFort III combined with a LeFort I advancement, each of 10 mm, for a 20 mm combined advancement. Stable, functional occlusion was achieved without airway compromise. This novel use of the combined LeFort III/I can restore stable class I occlusion in patients with BWS at risk for tongue base airway compromise.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-16T06:56:51Z
      DOI: 10.1177/10556656221101776
       
  • Assessment of the Velopharyngeal Mechanism at Rest and During Speech in
           Children With 22q11.2DS: A Cross-Sectional Study

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      Authors: Lakshmi Kollara, Jamie L. Perry, Richard E. Kirschner, Xiangming Fang, Adriane L. Baylis
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveVelopharyngeal dysfunction (VPD) associated with 22q11.2 deletion syndrome (22q11.2DS) has a complex etiology. This study had 3 aims: (1) assess differences in velopharyngeal and levator muscle configuration during rest versus sustained speech production (2) compare differences in velopharyngeal changes between children with and without 22q11.2DS (3) examine the relationship between adenoid thickness, pharyngeal depth, and velopharyngeal changes.DesignCross-sectional.MethodsA total of 22 participants, 11 with 22q11.2DS and 11 controls with normal speech and velopharyngeal anatomy (ages 4-12 years), underwent nonsedated MRI at rest and during sustained /i/. Differences in velar and levator muscle contraction across the 2 different conditions were analyzed, using matched paired t-tests. Mean differences across participant groups were examined. Correlation analyses were also conducted.ResultsWhen comparing differences between rest and sustained phoneme production (aim 1), significant (P < .05) differences were noted for all velar and levator muscle variables. For differences in velopharyngeal changes between children with and without 22q11.2DS (aim 2), VP ratio and effective VP ratio were noted to be significantly different. Pharyngeal depth and adenoid thickness were correlated with velar and levator muscle change measures and ratios (aim 3).ConclusionResults from this study provide quantitative in vivo measurements of the contracted levator muscle and velum in young children with 22q11.2DS. Results demonstrated that VP ratio and EVP ratio are significantly different between children with and without 22q11.2DS and that pharyngeal depth is a strong clinical determinant of VPD in children with 22q11.2DS.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-16T06:56:44Z
      DOI: 10.1177/10556656221100674
       
  • UCSQ Method Applied on 3D Photogrammetry: Non-Invasive Objective
           Differentiation Between Synostotic and Positional Plagiocephaly

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      Authors: Sophia A.J. Kronig, Otto D.M. Kronig, Henri A. Vrooman, Léon N.A. Van Adrichem
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveObjective differentiation between unilateral coronal synostosis (UCS) and positional posterior plagiocephaly (PPP) based on 3D photogrammetry according to Utrecht Cranial Shape Quantificator (UCSQ).DesignRetrospective study.SettingPrimary craniofacial center.Patients, ParticipantsThirty-two unoperated patients (17 UCS; 15 PPP) (age < 1 year).InterventionsExtraction of variables from sinusoid curves derived using UCSQ: asymmetry ratio forehead and occiput peak, ratio of gradient forehead and occiput peak, location forehead and occiput peak.Main Outcome Measure(s)Variables, derived using 3D photogrammetry, were analyzed for differentiation between UCS and PPP.ResultsFrontal peak was shifted to the right side of the head in left-sided UCS (mean x-value 207 [192-220]), and right-sided PPP (mean x-value 210 [200-216]), and to the left in right-sided UCS (mean x-value 161 [156-166]), and left-sided PPP (mean x-value 150 [144-154]). Occipital peak was significantly shifted to the right side of the head in left-sided PPP (mean x-value 338 [336-340]) and to the left in right-sided PPP (mean x-value 23 [14-32]). Mean x-value of occipital peak was 9 (354-30) in left- and 2 (350-12) in right-sided UCS. Calculated ratio of gradient of the frontal peak is, in combination with the calculated asymmetry ratio of the frontal peak, a distinctive finding.ConclusionsUCSQ objectively captures shape of synostotic and positional plagiocephaly using 3D photogrammetry, we therefore developed a suitable method to objectively differentiate UCS from PPP using radiation-free methods.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-11T07:20:53Z
      DOI: 10.1177/10556656221100679
       
  • The Clinical Significance of Clinocephaly in Late-Presentation Sagittal
           Craniosynostosis

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      Authors: Miles J. Pfaff, Regina Fenton, Aditya Mittal, Joseph W. Mocharnuk, Maryanna S. Owoc, Madeleine K. Bruce, Justin W. Beiriger, Joseph E. Losee, Jesse A. Goldstein
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundThe diagnosis of late-presentation sagittal suture craniosynostosis (SCS) can be challenging, especially in the setting of subtle physical exam findings. The clinical significance of clinocephaly—a retro-coronal concavity along the midvault—in this context remains unknown. The aim of this study is to evaluate the predictive value of clinocephaly in identifying late-presentation SCS.MethodsA retrospective chart review of all patients>1 year old presenting to the craniofacial clinic with a concern for SCS was performed. The presence or absence of SCS in the setting of clinocephaly was recorded following diagnostic imaging. Student's t test, Chi Square test, and multivariate logistic regression analysis were performed to determine predictors for SCS.Results75 patients met inclusion criteria. 32 patients (42.7%, 6% female) were diagnosed with SCS. No difference in age between patients with and without SCS was detected. Stratification of patients by age (1–2, 2–4, and>4 years) revealed a higher rate of SCS in younger patients (P = 0.04). The cephalic index (C.I.) of those with sagittal synostosis was significantly smaller but within the normal range, indicating a more scaphocephalic shape (P = 0.003). Logistic regression analysis revealed that C.I. was a strong predictor for SCS (P = 0.003). Of those with SCS, a mix of complete and partial fusion of the sagittal suture was appreciated.ConclusionThis study found that 42.7% of patients with clinocephaly had SCS. C.I. was the only predictor for SCS and unique suture fusion patterns were identified in those with SCS. This study suggests that clinocephaly should be considered a core component of the exam and work-up for SCS. Future studies aimed at evaluating the positive predictive value of this exam finding and identifying risk factors associated with late-presentation SCS are underway.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-11T07:20:25Z
      DOI: 10.1177/10556656211064484
       
  • What is the Effectiveness of Premaxilla Surgical Repositioning and its
           Stabilization Methods in Mixed Dentition Patients With Bilateral Cleft Lip
           and Palate'

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      Authors: Henrique de Vicq Normande Neto, Olavo Barbosa Oliveira-Neto, Igor Lerner Hora Ribeiro, Clarisse Samara de Andrade, Pedro Henrique da Hora Sales, Fernando José Camello de Lima
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveVerify the effectiveness of surgical repositioning of the premaxilla and its stabilization methods in patients with bilateral cleft lip and palate during mixed dentition.DesignSystematic review.Material and MethodsThe search was conducted in 7 databases (eg, Medline via PubMed; Scopus; Central Cochrane; LILACS; Embase, Web of Science; and Sigle via OpenGrey until August 2021), using the descriptors “premaxilla”, “cleft Palate”, and “bone transplantation”.Inclusion criteriaClinical trials and observational studies that have patients with bilateral cleft who had a need for superior/posterior repositioning of the premaxilla on mixed dentition; Studies in any language was evaluted whitout time restriction of publication.ResultsFrom 5572 records, 6 studies were included in the review with a total sample of 212 patients. Regarding the type of stabilization used in the premaxilla, the hybrid method (rigid and complementary semi-rigid stabilization) predominated, being observed in 184 patients (86.8%). A total of 17 failures were identified related to the surgical repositioning of the premaxilla, corresponding to 8% of the total number of surgeries. A meta-analysis of prevalence was performed, only with the retrospective studies. It was observed that the effectiveness rate of premaxilla repositioning was 92%, with a CI between 0.04 and 0.13, with all included studies showing a similar failure rate (0.08-0.09). The included studies also showed great homogeneity in this analysis (I2 = 0%; P = .75).ConclusionAlthough there are several alternatives and techniques for repositioning and stabilizing the premaxilla, the statistical result did not differ between the different techniques.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-10T06:16:39Z
      DOI: 10.1177/10556656221096304
       
  • Cephalometric Soft Tissue Morphology of Adults With Unoperated Submucous
           Cleft Palate

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      Authors: Congcong Cao, Xue Xu, Heng Yin, Qian Zheng, Chao Xu, Bing Shi
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe aim of this study was to compare craniofacial soft tissue characteristics between subjects with unrepaired submucous cleft palate (SMCP) and noncleft individuals.DesignThis retrospective cross-sectional study was performed on 27 subjects with unrepaired SMCP (13 male and 14 female subjects; mean age, 21.77 ± 4.09 years) and 30 noncleft controls (14 male and 16 female subjects; mean age, 22.67 ± 4.28 years). The predictor variable was cleft deformity. The outcome variable was cephalometric soft tissue measurements. Other study variables were gender and age. Independent samples t test and Mann-Whitney U test were used for intergroup comparison. P value was set at .05.ResultsSignificant differences were observed in the facial profile angle, total facial profile angle, soft tissue A-N-B angle, nasal base prominence, upper lip length, lower lip protrusion, and the ratio of upper lip length to mentolabial height between subjects with unoperated SMCP and noncleft controls.ConclusionsThe primary deformity of the cleft palate leads to unsatisfactory facial soft tissue morphology, especially in the middle facial region.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-09T11:07:13Z
      DOI: 10.1177/10556656221100125
       
  • The First Hybrid International Educational Comprehensive Cleft Care
           Workshop

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      Authors: Rami S. Kantar, Elçin Esenlik, Omar S. Al Abyad, Antonio Melhem, Robert A. Younan, Mario Haddad, Kristen Keith, Serena Kassam, Beyhan Annan, Charanya Vijayakumar, Arnaud Picard, Bonnie L. Padwa, Brian Sommerlad, Cassio Eduardo Raposo-Amaral, Christopher R. Forrest, David A. Gillett, Derek M. Steinbacher, Christopher M. Runyan, Daniela Y. S. Tanikawa, David K. Chong, David M. Fisher, Hans Mark, Halil Ibrahim Canter, Joseph E. Losee, Krishna G. Patel, Larry D. Hartzell, Adam B. Johnson, Marcus Vinícius Martins Collares, Nivaldo Alonso, Philip Kuo-Ting Chen, Raymond Tse, Robert J. Mann, Jose Rolando Prada-Madrid, Shinji Kobayashi, Syed Altaf Hussain, Ann Kummer, Debbie A. Sell, Valerie J. Pereira, Kelly Mabry, Courtney K. Gonsoulin, Martin Persson, Gareth Davies, Navil F. Sethna, Jennifer C. Munoz-Pareja, Anne Marie Kuijpers-Jagtman, Barry H. Grayson, Bruno Grollemund, Daniela G. Garib, Maria Costanza Meazzini, Om P. Kharbanda, Pedro E. Santiago, Prasad Nalabothu, Puneet Batra, Erin Stieber, Dushyant Prasad, Hugh Brewster, Ruben Ayala, Elif Erbay, M. Okan Akcam, J. Peter W. Don Griot, Raj M. Vyas, Roberto L Flores, Corstiaan C. Breugem, Usama S. Hamdan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveDescribe the first hybrid global simulation-based comprehensive cleft care workshop, evaluate impact on participants, and compare experiences based on in-person versus virtual attendance.DesignCross-sectional survey-based evaluation.SettingInternational comprehensive cleft care workshop.ParticipantsTotal of 489 participants.InterventionsThree-day simulation-based hybrid comprehensive cleft care workshop.Main Outcome MeasuresParticipant demographic data, perceived barriers and interventions needed for global comprehensive cleft care delivery, participant workshop satisfaction, and perceived short-term impact on practice stratified by in-person versus virtual attendance.ResultsThe workshop included 489 participants from 5 continents. The response rate was 39.9%. Participants perceived financial factors (30.3%) the most significant barrier and improvement in training (39.8%) as the most important intervention to overcome barriers facing cleft care delivery in low to middle-income countries. All participants reported a high level of satisfaction with the workshop and a strong positive perceived short-term impact on their practice. Importantly, while this was true for both in-person and virtual attendees, in-person attendees reported a significantly higher satisfaction with the workshop (28.63 ± 3.08 vs 27.63 ± 3.93; P = .04) and perceived impact on their clinical practice (22.37 ± 3.42 vs 21.02 ± 3.45 P = .01).ConclusionHybrid simulation-based educational comprehensive cleft care workshops are overall well received by participants and have a positive perceived impact on their clinical practices. In-person attendance is associated with significantly higher satisfaction and perceived impact on practice. Considering that financial and health constraints may limit live meeting attendance, future efforts will focus on making in-person and virtual attendance more comparable.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-05-09T11:06:48Z
      DOI: 10.1177/10556656221097820
       
  • Racial Disparities in the Timing of Alveolar Bone Grafting

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      Authors: Darin Patmon, Anna Carlson, John Girotto
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Standard bone grafting between ages 6 and 12 has become the preferred treatment of choice for alveolar clefts. Given the importance of surgical timing in complete cleft palate repairs, it is important to identify any populations at-risk for delayed alveolar bone grafting. The purpose of this study is to identify whether a racial disparity is present nationally in the timing of alveolar bone grafting.Design:Retrospective analysis Setting: Hospitals participating in the American College of Surgeons National Surgical Quality Improvement Program (ACS-NSQIP).Patients:Inclusion criteria involved patients who underwent alveolar bone grafting identified by current procedure terminology (CPT) code 42210 between years 2012 and 2019. Patients were stratified by age at time of operation based on the following parameters: early bone grafting (before 6 years of age), standard bone grafting (between 6 and 12 years of age), and late bone grafting (after 12 years of age).Main Outcome Measures:Racial and ethnic differences in the age of patients at the time of alveolar bone grafting.Results:Overall, 20.28% of the cohort received alveolar bone graft after 12 years of age. African American (29.33%) and Hispanic (24.42%) patients received late alveolar bone grafting more frequently than other racial and ethnic groups (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-28T07:54:59Z
      DOI: 10.1177/10556656221097813
       
  • Novel Digital Workflow for Nasoalveolar Molding and Postoperative Nasal
           Stent for Infants With Cleft Lip and Palate

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      Authors: Chad B. Carter, Francisco F. Gallardo, Hannah E. Colburn, Daniel W. Schlieder
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveWe present a novel digital workflow to provide presurgical infant orthopedic (PSIO) treatment for a patient with a unilateral cleft lip/palate utilizing nasoalveolar molding (NAM) and a custom postsurgical nasal stent.SettingWithin the US military healthcare system, the Joint Base San Antonio Craniofacial Anomalies Team utilizes dental scanners, predictive 3D modeling software, and 3D printing technology in a digital workflow for NAM appliance fabrication.WorkflowSoft tissue facial scanning, peri-oral scanning, and dental putty impressions are used to facilitate fabrication and measure outcomes. Digital modeling software and 3D resin printing are utilized to manufacture the prescribed devices.Main Outcome Measures and ResultsExtra-oral facial scans and intra-oral impressions are compared between 3 timepoints: pre-treatment, posttreatment with NAM, and postsurgical treatment.ConclusionsThe ability to share workflows, establish outcome standards, and streamline patient care will continue to advance best practices in digital PSIO.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-28T07:49:03Z
      DOI: 10.1177/10556656221095393
       
  • Formula Otorrhea in Pediatric Patients With Cleft Palate

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      Authors: Tyler J. Gathman, Janet S. Choi, Brianne B. Roby, Tina Huang
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      The case series details 2 unusual cases of male newborns with cleft lip and palate (CLP) that later developed formula otorrhea. Both patients underwent bilateral myringotomies with the insertion of pressure equalizing (PE) tubes for chronic otitis media with effusion (OME). Chronic otorrhea associated with feeding occurred post-PE tube insertion and the otorrhea was later confirmed to be due to reflux of formula. Patients were treated with antibiotic ear drops, routine ear cleaning, anti-reflux medication, and reflux precautions. After definite cleft palate repair, formula otorrhea completely resolved. When patients with CLP develop chronic OME or otorrhea following PE tube placement, reflux of formula into the middle ear should be considered and treated accordingly.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-28T03:37:16Z
      DOI: 10.1177/10556656221097822
       
  • Neck Extension and Intraocular Pressure Elevation During Palatoplasty in a
           Patient with Patau Syndrome and Congenital Glaucoma: A Case Report

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      Authors: Jihion Yu, Jae Yong Kim, Jiwoong Lee, Jun-Young Park, Jai-Hyun Hwang, Tae Suk Oh, Woo Shik Jeong, Young-Kug Kim
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Patau syndrome (trisomy 13) is a severe disorder associated with multiple systemic defects. Patau syndrome is commonly associated with ocular abnormalities but rarely associated with congenital glaucoma. To obtain a better surgical view, palatoplasty requires neck extension during surgery. The intraocular pressure (IOP) of patients with Patau syndrome can increase owing to the neck extension position while undergoing palatoplasty, particularly in those with congenital glaucoma. Here, we describe a case with increased IOP measured using a rebound tonometer during palatoplasty in a pediatric patient with Patau syndrome and congenital glaucoma. This case shows that it may be important to reduce the degree of neck extension and shorten the operation time to minimize any increase in the IOP during palatoplasty in pediatric patients with Patau syndrome accompanied by congenital glaucoma.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-27T07:46:55Z
      DOI: 10.1177/10556656221097209
       
  • Proboscis Lateralis With Basal Encephalocele: A Report of Clinical
           Management and Reconstructive Approach

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      Authors: Sarut Chaisrisawadisuk, Inthira Khampalikit, Mark H. Moore, Peter J. Anderson, Somboon Chaisrisawadisuk
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Proboscis lateralis is a rare craniofacial anomaly in which a rudimentary nasal appendage arises at the medial canthal area. The severity depends on organ involvement, including eyes, nose, cleft lip/palate, and/or concomitant intracranial anomalies. Here, we present a child with proboscis lateralis and associated trans-ethmoidal encephalocele. We suggest doing the preoperative CT and/or MRI to rule out associated intracranial anomalies and reliably preoperative planning tools. Moreover, we proposed an alternative nasal reconstructive technique using a composite graft from the proboscis mass at the same time as encephalocele repair with promising results.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-27T07:46:36Z
      DOI: 10.1177/10556656221096323
       
  • Evaluating International Diagnostic, Screening, and Monitoring Practices
           for Craniofacial Microsomia and Microtia: A Survey Study

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      Authors: Elsa M. Ronde, Jitske W. Nolte, Frea H. Kruisinga, Saskia M. Maas, Oren Lapid, Fenna A. Ebbens, Alfred G. Becking, Corstiaan C. Breugem
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivesTo (1) appraise current international classification and clinical management strategies for craniofacial microsomia (CFM) and microtia, and (2) to assess agreement with the European Reference Network “European Guideline Craniofacial Microsomia” recommendations on screening and monitoring.DesignThis was a cross-sectional online survey study. The survey consisted of 44 questions on demographics, diagnostics and classification, obstructive sleep apnea, feeding difficulties, speech and language development, hearing, ocular abnormalities, visual development, orthodontic screening, genetic counselling, psychological wellbeing, and extracraniofacial anomalies.ParticipantsRespondents were participants of 3 international cleft and craniofacial conferences, members of the American Cleft Palate and Craniofacial Association and members of the International Society for Auricular Reconstruction. Respondents were requested to complete 1 questionnaire per multidisciplinary team.ResultsFifty-seven responses were received from 30 countries (response rate ∼3%).The International Consortium for Health Outcomes Measurement diagnostic criteria were used by 86% of respondents, though 65% considered isolated microtia a mild form of CFM. The Orbit, Mandible, Ear, Facial Nerve and Soft Tissue classification system was used by 74% of respondents. Agreement with standardized screening and monitoring recommendations was between 61% and 97%. A majority of respondents agreed with screening for extracraniofacial anomalies (63%-68%) and with genetic counselling (81%).ConclusionsThis survey did not reveal consistent agreement on the diagnostic criteria for CFM. Respondents mostly supported management recommendations, but frequently disagreed with the standardization of care. Future studies could focus on working towards international consensus on diagnostic criteria, and exploring internationally feasible management strategies.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-26T07:23:53Z
      DOI: 10.1177/10556656221093912
       
  • Implementation of an Ambulatory Cleft Lip Repair Protocol: Surgical
           Outcomes

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      Authors: Jenn J. Park, Ricardo Rodriguez Colon, Bachar F. Chaya, Danielle H. Rochlin, Patricia D. Chibarro, Pradip R. Shetye, David A. Staffenberg, Roberto L. Flores
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivesCleft lip repair has traditionally been performed as an inpatient procedure. There has been an interest toward outpatient cleft lip repair to reduce healthcare costs and avoid unnecessary hospital stay. We report surgical outcomes following implementation of an ambulatory cleft lip repair protocol and hypothesize that an ambulatory repair results in comparable safety outcomes to inpatient repair.Design/SettingThis is a single-institution, retrospective study.Patients/ParticipantsPatients undergoing primary unilateral (UCL) and bilateral (BCL) cleft lip repair from 2012 to 2021 with a minimum 30-day follow-up. A total of 226 patients with UCL and 58 patients with BCL were included.InterventionAmbulatory surgery protocol in 2016.Outcome MeasuresVariables include demographics and surgical data including 30-day readmission, 30-day reoperation, and postoperative complications.ResultsThere were no differences in rates of 30-day readmission, reoperation, wound complications, or postoperative complications between the pre- and post-protocol groups. Following ambulatory protocol implementation, 80% of the UCL group and 56% of the BCL group received ambulatory surgery. Average length of stay dropped from 24 h pre-protocol to 8 h post-protocol. The 20% of the UCL group and 44% of the BCL group chosen for overnight stay had a significantly higher proportion of congenital abnormalities and higher American Society of Anesthesiology (ASA) class. Reasons for overnight stay included cardiac/airway monitoring, prematurity, and monitoring of comorbidities. There were no differences in surgical outcomes between the ambulatory and overnight stay groups.ConclusionsAn ambulatory cleft lip repair protocol can significantly reduce average length of stay without adversely affecting surgical outcomes.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-26T06:51:03Z
      DOI: 10.1177/10556656221096567
       
  • Orthodontic Treatment of Patients With Clefts: Satisfaction and
           Psychological Aspects

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      Authors: Olga Forer, Erica Cohen, Yocheved Ben-Bassat, Avraham Zini, Miriam Shalish
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo assess parents’ satisfaction with cleft child's facial appearance and function; compare these findings with orthodontist (expert) satisfaction and evaluate influence of various factors on satisfaction.DesignCross-sectional study.MethodsSixty-three parents of non-syndromic patients with clefts (ages 7-20 years), and an orthodontist, completed the Cleft Hearing, Appearance and Speech Questionnaire. Two scores were produced: cleft-associated, and non-cleft-associated features. Additional open questions were presented to the parents.ResultsBoth parents and orthodontist gave high satisfaction scores (mean: 8.4 & 8.2, respectively), significantly correlated, for the cleft-associated features (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-26T06:50:33Z
      DOI: 10.1177/10556656221093934
       
  • COVID-19 Pandemic Associated With Increased Self-reported Depressive
           Symptoms in Patients With Congenital Craniofacial Diagnoses

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      Authors: Kelly X. Huang, Michelle K. Oberoi, Rachel M. Caprini, Vivian J. Hu, Sri Harshini Malapati, Sarah Mirzaie, Meiwand Bedar, Harsh Patel, Justine C. Lee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe current study investigated the influence of the coronavirus (COVID-19) pandemic on patients with congenital craniofacial diagnoses.MethodsPatients (n = 66) with craniofacial diagnoses aged between 8 and 17 were prospectively evaluated with longitudinal psychosocial assessments using the anger, anxiety, depressive symptoms, and peer relationships instruments within the pediatric Patient-Reported Outcomes Measurement Information System (PROMIS). The COVID-19 cohort (n = 33) included patients with assessments within 2 years prior to the pandemic (t0) and during the pandemic (t1; March 2020 to March 2021). An age-matched comparison cohort (n = 33) with similar demographics and diagnoses included patients assessed twice over 3 years prior to the pandemic.ResultsAll PROMIS measures were in the average range clinically for both groups across time points. However, the COVID-19 group reported a significant increase in depressive symptoms during the pandemic (t1) compared to pre-pandemic (t0) scores (48.2 ± 10.1 vs 44.3 ± 9.4, P = .04, d = −0.37), while the comparison group did not demonstrate any differences in psychosocial functioning between t0 and t1. For the COVID-19 cohort, only the pandemic timeframe (r = 0.21, P = .03) was significantly associated with increased depressive symptom scores, and no other sociodemographic or medical variables were associated with depressive symptoms.ConclusionsSelf-reported depressive symptoms increased during the COVID-19 pandemic in patients with congenital craniofacial diagnoses. Longitudinal studies are needed to elucidate whether such changes will be persistent or compound known variables associated with psychosocial functioning.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-26T06:50:23Z
      DOI: 10.1177/10556656221095715
       
  • Occlusal Evaluation Using Modified Huddart and Bodenham Scoring System
           Following 2-Stage Palatoplasty With Hotz Plate: A Comparison Among 3
           Different Surgical Protocols

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      Authors: Andrea Rei Estacio Salazar, Yasumitsu Kodama, Ryutaro Yuki, Rei Ominato, Takahiro Nagai, Momoko Watanabe, Akane Yamada, Ryota Kobayashi, Kaya Ichikawa, Jun Nihara, Akihiko Iida, Kazuhiro Ono, Isao Saito, Ritsuo Takagi
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aimed to determine if the change in technique of soft palate closure or timing of hard palatal repair induced occlusal changes in patients with complete unilateral cleft lip and palate (CUCLP).DesignRetrospective study.SettingsA medical and dental hospital in Japan.SubjectsA total of 96 patients with CUCLP treated with 2-stage palatoplasty were included in the study and categorized into 3 groups (G1, G2, and G3) according to the protocol used.InterventionsG1 underwent soft palate repair using Perko method at 1.5 years of age and hard palate repair using vomer flap procedure at 5.5 years of age. Furlow method was used for soft palate repair in G2 at 1.5 years of age and hard palate repair using vomer flap procedure at 5.5 years of age. The Furlow method was used to repair the soft palate in G3 at 1.5 years of age and vomer flap procedure was used to repair the hard palate at 4 years of age.Main Outcome MeasuresTwo evaluators assessed the dental arch relationship using the modified Huddart/Bodenham (mHB) index on 2 separate occasions.ResultsIntra- (intraclass correlation coefficient [ICC]: 0.962) and inter-examiner (ICC: 0.950) reliability showed very good agreement. The frequency of crossbite present in the major and minor segments gradually decreased with each change in protocol. Mean segmental scores showed no significant difference between 3 protocols (P > .05). Good inter-arch alignment occurred with all 3 surgical protocols (G1:82.6%, G2:89.8%, and G3:91.7%).ConclusionsThere was no significant difference in the dental arch relationship outcomes between the 3 surgical protocols. The dentition status was comparable with all surgical protocols, even after the changes.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-26T06:50:14Z
      DOI: 10.1177/10556656221093293
       
  • Customized Tray for Impression Taking in Children With Cleft Lip and
           Palate

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      Authors: Cleide Felício Carvalho Carrara, Paula Karine Jorge, Beatriz Costa, Maria Aparecida Andrade Moreira Machado, Thais Marchini Oliveira, Gisele da Silva Dalben
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      The assessment of rehabilitation outcomes requires a patient documentation protocol, including records obtained at standardized ages, to compare different types of surgeries, their effects, as well as between different rehabilitation centers. The aim of this paper was to present proper trays for babies with different types of cleft lip and palate, which are used in the outpatient routine at Hospital of Rehabilitation of Craniofacial Anomalies/USP (HRAC/USP). The customized trays are made with self-curing acrylic resin. The tray must have suitable depth to copy the buccal sulcus, and wax is usually applied to contour the tray edge, and the adjustment of the tray to the fornix, making the tray specific for each child. The impression precludes the utilization of dental casts for diagnosis, treatment plan, and research measurements. In the clinical practice at HRAC-USP, it was observed that customized trays increased the quality of impression, accurately reproducing anatomical features of dental arches of babies with oral clefts.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-25T06:02:50Z
      DOI: 10.1177/10556656221095713
       
  • Salvage of Ear Framework Exposure Following Autologous Microtia
           Reconstruction: Repair Strategy for Each Location of Exposure

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      Authors: Masaki Fujioka, Kiyoko Fukui, Kentaro Yoshino, Miho Noguchi, Ryuichi Murakami
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      One of the most common complications of total auricular reconstruction is exposure of the ear framework. Various reconstruction methods have been reported depending on the location and size of exposed cartilage. This report describes a safe reconstruction method for each exposed part of the grafted ear framework. From January 2019 to August 2021, 2 cases (4 areas) of framework exposure were observed following autologous microtia reconstruction. The first case developed 2 small areas of skin necrosis on the anterior helix and lower antihelix to concha. The former was reconstructed with a temporal fascia flap and the latter with a local transposition flap. The second case also developed 2 small areas of skin necrosis on the posterior helix and lower antihelix to concha. The former was sutured directly and the latter with a local transposition flap. However, both wounds recurred due to flap necrosis and the cartilage was exposed again. The 3rd operation was performed by covering both wounds with a posterior auricular turnover flap and skin graft. In both cases, the exposed framework was completely covered with the flaps, and the reconstructed ears showed well-defined convolutions. Covering exposed cartilage with a local flap with a random pattern of blood circulation is convenient because no additional skin grafts are required. However, the blood circulation of the flaps is inadequate when an elongated flap is required; consequently, flap necrosis may occur. On the other hand, a temporal fascia flap and posterior auricular flap, which have axillary pattern blood circulation, are considered to be safer. We believe that it is safe to use a temporal fascia flap for cartilage exposure in the upper half of the auricle, and a posterior auricular turnover flap for the lower half.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-22T06:16:39Z
      DOI: 10.1177/10556656221095389
       
  • Design of a Novel Orthodontic Appliance to Prevent Pedicle Trauma in
           Patients Undergoing Double-Opposing Buccal Flaps for Palatal Lengthening

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      Authors: Sarah E. Hill, Ilana M. Ickow, Waverley He, Rae A. Buckley, Jordan P. Steinberg
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo describe a novel orthodontic appliance to prevent pedicle trauma in patients undergoing double-opposing buccal flap surgery for secondary palatal lengthening.DesignCase series.SettingCleft and craniofacial clinic, Johns Hopkins Children's Center.Patients, ParticipantsFour patients undergoing double-opposing buccal flap surgery for repair of velopharyngeal insufficiency.InterventionsPatients were fitted with the device, which consists of a lower lingual holding arch with acrylic bite blocks.Main Outcome MeasurePresence of pedicle trauma postsurgery and tolerability of the device.ResultsThe appliance was well tolerated in all 4 patients and no biting trauma to the pedicles was observed.ConclusionsA reliable appliance has been developed to prevent biting trauma to the pedicles in patients undergoing double-opposing buccal flap surgery in the permanent dentition stage.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-22T06:15:48Z
      DOI: 10.1177/10556656211069836
       
  • Application of Computer-Assisted Preoperative Planning in Treating
           Congenital Bilateral Zygomatico-Maxillo-Mandibular Fusion: A Case Report
           and Literature Review

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      Authors: Sheng Chen, Jie Cui, Yi Ji, Liangliang Kong, Weimin Shen
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Congenital fusion of the jaws (syngnathia) is a rare facial malformation with an unknown etiology. This disease may vary in severity with adhesion of soft tissue and bony fusion. It can be anterior fusion, unilateral or bilateral fusion, and complete fusion. The main problem of these patients is the difficulty of airway maintenance and feeding, and the most common postoperative complication is the relapse of bony fusion. Here, we report a young male patient with bony syngnathia, involving bilateral fusion of the ascending ramus and body of the mandible with the maxillary complex. We performed bone isolation by computer-assisted preoperative planning and used an insertional temporalis flap to fix the wound surface to prevent refusion of bone.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-20T06:21:27Z
      DOI: 10.1177/10556656211064775
       
  • Narcotic Utilization After Cleft Lip Repair: Does Local Anesthetic Choice
           Matter'

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      Authors: Christopher L Kalmar, Zachary D Zapatero, Mychajlo S Kosyk, Jordan W Swanson, Jesse A Taylor
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo analyze whether the choice of intraoperative local anesthetic for cleft lip repair is associated with the amount of perioperative narcotic utilization.DesignRetrospective cohort study.SettingHospitals participating in the Pediatric Health Information System.PatientsPrimary cleft lip repairs performed in the United States from 2010 to 2020.InterventionsLocal anesthesia injected—treatment with lidocaine alone, bupivacaine alone, or treatment with both agents.Outcome MeasuresPerioperative narcotic administration.ResultsDuring the study interval, 8954 patients underwent primary cleft lip repair. Narcotic utilization for unilateral (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-19T05:57:59Z
      DOI: 10.1177/10556656221093945
       
  • Prenatal Consultation Outcomes for Infants With Cleft Lip With and Without
           Cleft Palate

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      Authors: Rachel L. Thompson, Heidi L. Thorson, Sivakumar Chinnadurai, Robert J. Tibesar, Brianne B. Roby
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo assess the clinical impacts of prenatal consultation with a multidisciplinary cleft team on infants with cleft lip with or without cleft palate (CL ± P).DesignRetrospective cases series.SettingTertiary pediatric hospital.PatientsInfants with CL ± P whose mothers received prenatal consultation with a pediatric otolaryngology team from June 2005 to December 2019 were identified. A random sample of infants with CL ± P without prenatal consultation from June 2005 to December 2019 was also identified.Main Outcome MeasuresThe primary outcomes were the length of hospitalization during the first 12 weeks of life, timing of surgical repair, length of postsurgical hospitalization, and number of unplanned clinic visits and phone calls for feeding evaluation.ResultsTime to cleft lip repair differed significantly between the 2 groups with repair performed at 13.4 (±0.9) weeks for the prenatal consultation group (n = 73) and 15.3 (±2.1) weeks for the control group (n = 80), (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-19T05:57:50Z
      DOI: 10.1177/10556656221093174
       
  • Evaluation of Facial Esthetics Following NAM Versus CAD/NAM in Infants
           With Bilateral Cleft Lip and Palate: A Randomized Clinical Trial

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      Authors: Noha A. El-Ashmawi, Mona M. Salah Fayed, Amr El-Beialy, Ahmed E. Fares, Khaled H. Attia
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe aim is to compare the facial esthetics following nasoalveolar molding (NAM) versus computer-aided design NAM (CAD/NAM) in patients with bilateral cleft lip/palate (CLP).DesignThe trial is a randomized comparative trial with a 1:1 allocation ratio.ParticipantsThirty infants with bilateral complete cleft lip and palate were recruited.InterventionsPatients were randomized between NAM and CAD/NAM groups. The treatment steps described by Grayson were followed for the NAM group. In the CAD/NAM group, digitized maxillary models were made to create a series of modified virtual models, which were used to fabricate the molding plates using 3-dimensional printing technology. The nasal stents were then added to the intraoral plates following the Grayson method. The study lasted for 4 months.Main OutcomesThe assessment of the changes observed in the interlabial gap and nasolabial esthetics was done using standardized 2-dimensional photographs. The correlation between dental arch changes and extraoral facial esthetics was studied.ResultsBoth modalities showed improvement in nasolabial esthetics before the lip surgery. No statistically significant difference was found between NAM and CAD/NAM groups in any of the assessed variables.ConclusionsBoth interventions were effective in the management of infants with bilateral CLP.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-15T06:03:51Z
      DOI: 10.1177/10556656221093176
       
  • Assessing Craniofacial Dysmorphology and Postoperative Outcomes: Past
           Perils and Promising Pearls in Plastic Surgery

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      Authors: Dillan F. Villavisanis, Scott P. Bartlett, Jesse A. Taylor
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.

      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-15T05:43:10Z
      DOI: 10.1177/10556656221093911
       
  • Three-Dimensional Assessment of Eustachian Tube in Patients With Cleft
           Palate Versus Controls: A CBCT Study

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      Authors: Mahvash Hasani, Shabnam Ajami, Golnoush Farzinnia
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo compare the morphological characteristics of the Eustachian tube (ET) between patients with cleft palate and healthy controls with the use of cone-beam computed tomography (CBCT).DesignRetrospective assessment of treated nonsyndromic patients with cleft palate and a control group.ParticipantsCBCT images of 51 patients with cleft palate who had previously undergone surgery to close the cleft palate and a control group of 51 patients were included in this study. Syndromic patients were excluded.MethodsThe cleft group were divided into 3 groups according to the palatoplasty technique, which was done in the infancy period to close the cleft palate (Nadjmi modification of Furlow, Sommerlad, and V-Y pushback). Finally, ET length (ETL), ET angle, and auditory tube angle (ATA) were measured in all patients.ResultsThe ETL, ETA, and ATA in the cleft and the normal control groups were 29.73 ± 3.14 and 32.18 ± 3.34 mm, 33.18 ± 2.57 and 35.40 ± 3.93, and 141.64 ± 4.13 and 138.87 ± 4.96, respectively. All 3 features were statistically significant among the groups (All Ps 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-13T07:03:01Z
      DOI: 10.1177/10556656221093554
       
  • YouTube as a Source of Patient Education for Transcranial Craniosynostosis
           Procedures

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      Authors: Catrin Stallwood-Hall, Andrew May, Mark Moore
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionThe internet has improved access to knowledge, with a reported 72% of internet users accessing health information online. YouTube is the most popular video-sharing website and is a frequently accessed source for health information. There are concerns, however, that patients and carers may be unable to identify and filter for trustworthy sources of information on these platforms. This study aimed to assess the quality of videos that are likely to be accessed when families search YouTube for transcranial procedures for the management of craniosynostosis.MethodologyThis was a cross-sectional, observational study that evaluated YouTube videos when searching for “fronto-orbital advancement” or “cranial vault remodeling.” The first 25 videos for each term were included. Videos were assessed for quality using the previously validated JAMA and DISCERN scoring tools.ResultsThere was low interobserver variability for the scoring tools used. There was no statistically significant correlation between the video quality and any video characteristics assessed. Videos published by organizations had higher mean quality scores for both the JAMA and DISCERN tools when compared to surgeon, patient, or medical device company-produced videos.ConclusionThis study showed that searching YouTube for fronto-orbital advancement and cranial vault remodeling yields videos of low quality and therefore patients and carers should be discouraged from “YouTube-ing” these procedures. Platforms such as YouTube may provide an opportunity for craniofacial units to create and disseminate high-quality educational videos to complement routine perioperative education.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-11T03:07:18Z
      DOI: 10.1177/10556656221093292
       
  • Psychosocial and Health-Related Experiences of Individuals With Microtia
           and Craniofacial Microsomia and Their Families: Narrative Review Over 2
           Decades

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      Authors: Alexis L Johns, Nicola M Stock, Bruna Costa, Kristin Billaud Feragen, Canice E Crerand
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:This paper describes 20 years of microtia and craniofacial microsomia (CFM) psychosocial and healthcare studies and suggests directions for clinical care and research.Design:A narrative review of papers January 2000 to July 2021 related to psychosocial and healthcare experiences of individuals with microtia and CFM and their families.Results:Studies (N = 64) were mainly cross-sectional (69%), included a range of standardized measures (64%), and were with European (31%), American (27%), or multinational (23%) samples. Data were generally collected from both patients and caregivers (38%) or patient self-report (35%). Sample sizes were 11 to 25 (21%), 26 to 50 (19%), 51 to 100 (22%), or over 100 (38%). Studies addressed 5 primary topics: (1) Healthcare Experiences, including Medical Care, Hearing Loss/Amplification, Diagnostic Experiences, and Information Preferences; (2) Psychosocial Experiences, including Teasing, Behavioral Adjustment, Psychosocial Support, and Public Perception; (3) Neurocognitive Functioning and Academic Assistance; (4) Pre- and Post-Operative Psychosocial Outcomes of Ear Reconstruction/Canaloplasty; and (5) Quality of Life and Patient Satisfaction.Conclusions:Care involved multiple specialties and was often experienced as stressful starting at diagnosis. Psychosocial and neurocognitive functioning were generally in the average range, with possible risk for social and language concerns. Coping and resiliency were described into adulthood. Satisfaction and positive benefit of ear reconstruction/canaloplasty were high. Care recommendations include increasing: hearing amplification use, microtia and CFM knowledge among providers, efficient treatment coordination, psychosocial support, academic assistance, and advances to minimize surgical scarring. This broad literature overview informs clinical practice and research to improve psychosocial outcomes.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-06T05:57:23Z
      DOI: 10.1177/10556656221091699
       
  • Range and Frequency of Congenital Malformations Among Children With Cleft
           Lip and/or Palate

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      Authors: KJ Fitzsimons, MJ Hamilton, J van der Meulen, J Medina, MAH Wahedally, MH Park, CJH Russell
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo assess the range and frequency of additional congenital malformations identified among children born alive with CL/P.DesignAnalysis of patient-level data from a national registry of cleft births linked to national administrative data of hospital admissions.SettingNational Health Service, England.PatientsChildren born between 2000 and 2012 receiving cleft care in English NHS hospitals.Outcome MeasuresThe proportion of children with ICD-10 codes for additional congenital malformations, according to cleft type.ResultsThe study included 9403 children. Of these 2114 (22.5%) had CL±A, 4509 (48.0%) had CP, 1896 (20.2%) had UCLP, and 884 (9.4%) had BCLP. A total of 3653 (38.8%) children had additional congenital malformations documented in their hospital admission records. The prevalence of additional congenital malformations was greatest among children with CP (53.0%), followed by those with BCLP (33.5%), UCLP (26.3%), and then CL±A (22.2%) (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-06T05:57:16Z
      DOI: 10.1177/10556656221089160
       
  • Timing of Spheno-Occipital Synchondrosis Closure in Cleft Lip and Palate
           Patients in Iranian Population

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      Authors: Mahvash Hasani, Marzieh Mokhberi, Maryam Karandish, Rabieh Boroumand
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aimed to evaluate the chronological age range associated with each stage of spheno-occipital synchondrosis (SOS) fusion in patients with cleft lip and palate compared to the noncleft group, using cone-beam computed tomography (CBCT) images. Design: In this study, the degree of SOS fusion was assessed using a 4-stage scoring system on CBCT images of 190 individuals (92 patients with cleft lip and palate, 98 noncleft individuals). A χ2 test was performed to assess the correlation between age and fusion stage. The independent sample t tests were used to evaluate the differences in the mean values of the samples of each group and each sex, as well as the mean value of each stage (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-06T03:22:10Z
      DOI: 10.1177/10556656221091711
       
  • Co-Occurrence of Taurodontism in Nonsyndromic Cleft Lip and Palate
           Patients in Subset of Indian Population: A Case–Control Study Using CBCT
           

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      Authors: Geetika Sobti, Astha Chaudhry, Jaishree Thanvi, Isha Gaurav, Chandni Shekhawat, Debopriya Banerjee, Lalit Kumar Likhyani, Sugandhi Soni
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivesThe present study aimed to assess the co-occurrence of taurodontism in nonsyndromic cleft lip and palate (NSCLP) patients in a subset of Indian population using cone beam computed tomography (CBCT).MethodThe study was a retrospective case–control kinds assessing 1500 CBCT scans over a period of 2 years; 67 scans out of 1500 showed cleft lip and palate (CLP). After fulfilling the inclusion and exclusion criteria, 38 subjects out of 67 comprised the case group; 80 randomly selected subjects constituted the control group. The first and second permanent molars in both the arches (8 teeth) of each subject were assessed for the presence and severity of taurodontism using the objective criteria. Statistical analysis was done using the χ2 test. Inter and intraobserver agreement was evaluated by Kappa statistics.Results71.05% subjects showed taurodontism in case group, while 45% subjects showed its presence in control group; results being significant for both the groups. However, the comparison of number of teeth with taurodontism in the 2 groups came out to be statistically insignificant. Also no significant association of taurodontism was seen with gender or the different types of cleft (P = .437). Hypotaurodontism was the most prevalent type.ConclusionThere was a higher prevalence of taurodontism in both groups which had been underestimated in the previous studies. This emphasizes the significance of the use of CBCT in detection of dental anomalies which would otherwise go undetected.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-04T08:16:44Z
      DOI: 10.1177/10556656221089159
       
  • Parents’ Perception of Barriers to the Comprehensive Management of
           Children With Cleft Lip and Palate in Bogota, Colombia

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      Authors: María Clara González-Carrera, Jaime Alberto Ruiz, Ingrid Isabel Mora-Díaz, David Pereira de Souza, Luis Fernando Restrepo-Pérez, Zita Bendahan, Gloria Rocío Bautista, David Díaz-Báez
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectTo identify the perception of barriers to the comprehensive management of cleft lip and palate (CLP) by parents/caregivers of Colombian children with this condition.Setting and Sample PopulationFifty parents/caregivers of children with CLP under 12 years attending a center specialized in the management of craniofacial congenital conditions in Bogota, Colombia.Materials and MethodsThis study consisted of 2 phases: a quantitative phase (descriptive cross-sectional) and a qualitative phase (focus group [FG]). Chi-square and Fisher exact tests were used to analyze the association variables. The barriers and alternative ways to overcome barriers were analyzed in the FG.ResultsComprehensive management was mostly defined as access to multiple treatments (54%), and this concept was expanded in the FG toward understanding CLP at all levels. Monoparental families spend their income on treatments (29%) than nuclear families (0%) (P = .001). All parents with high education levels were familiar with healthcare centers specialized in CLP as opposed to 66.7% of parents with basic primary education (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-04-01T06:08:23Z
      DOI: 10.1177/10556656221082759
       
  • Postnatal Progressive Craniosynostosis in Syndromic Conditions: Two
           Patients With Saethre-Chotzen Due to TWIST1 Gene Deletions and Review of
           the Literature

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      Authors: Rama J Alawneh, Andrea L Johnson, Julie Elizabeth Hoover-Fong, Eric M Jackson, Jordan P Steinberg, Gretchen MacCarrick
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Saethre-Chotzen syndrome (SCS) is a known craniosynostosis syndrome with a variable presentation of craniofacial and somatic involvement. Congenital coronal craniosynostosis is most commonly observed in SCS; however, progressive postnatal craniosynostosis of other sutures has been reported. The authors present 2 infants with progressive postnatal craniosynostosis and SCS caused by chromosome 7p deletions including the TWIST1 gene. The evolution of their clinical features and a literature review of patients with syndromic, postnatal progressive craniosynostosis illustrate the importance of longitudinal observation and management of these patients.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-31T05:30:08Z
      DOI: 10.1177/10556656221090844
       
  • Third-party Compliance With State-Mandated Orthodontic Coverage for Cleft
           Palate and Craniofacial Care in California: An Insurance Claim Thematic
           Analysis

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      Authors: Ronnel Azizollahi, Laura Hubacek, Snehlata Oberoi, Cristin Kearns
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundCalifornia Senate Bill 630 (SB630) enacted statutorily mandated health plan coverage for orthodontic care of patients with cleft palate and craniofacial anomalies in 2009, which was effective from July 1, 2010. In this qualitative analysis, third-party compliance with SB630 in a university-based cleft and craniofacial orthodontic program is evaluated.MethodsPrivately insured patients that experienced a coverage delay or denial of orthodontic treatment for cleft lip and palate in the University of California, San Francisco Cleft and Craniofacial Orthodontic Program between July 1, 2010 and October 28, 2020 were identified. A thematic analysis of reasons for delay or denial was conducted.ResultsNearly three quarters of patients experienced coverage delay and/or denials. The most common reason given was that services were not covered.ConclusionsDespite state-mandated coverage, inappropriate denials of orthodontic care for patients with cleft lip and palate by private insurers persist in California.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-31T05:29:40Z
      DOI: 10.1177/10556656221074523
       
  • Multimodal Treatment of Robin Sequence Utilizing Mandibular Distraction
           Osteogenesis and Continuous Positive Airway Pressure

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      Authors: Mychajlo S. Kosyk, Anna R. Carlson, Zachary D. Zapatero, Christopher L. Kalmar, Sidra Liaquat, Scott P. Bartlett, Jesse A. Taylor, Christopher M. Cielo, Jordan W. Swanson
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveMandibular distraction osteogenesis (MDO) and continuous positive airway pressure (CPAP) may each have a role in effectively treating tongue-based airway obstruction (TBAO) in Robin sequence (RS). This study describes longitudinal outcomes after treatment of TBAO with CPAP and/or MDO.DesignRetrospective cohort study.SettingTertiary Pediatric Hospital.PatientsA total of 129 patients with RS treated with CPAP and/or MDO from 2009 to 2019 were reviewed. Subjects receiving baseline and at least one follow-up polysomnogram were included. 55 who underwent MDO ± CPAP and 9 who received CPAP-only treatment were included.Main Outcome MeasuresPatient characteristics, feeding, and polysomnographic data were compared and generalized linear mixed modeling performed.ResultsBaseline obstructive apnea–hypopnea index (OAHI) was greater in the MDO-treated group (median x˜ = 33.7 [interquartile range: 26.5-54.5] than the CPAP-treated group (x˜ = 20.3[13.3-36.7], P ≤ .033). There was significant reduction in OAHI following treatment with CPAP and MDO modalities, P ≤ .001. SpO2 nadir after MDO was lower in syndromic (x˜ = 85.0[81.0-87.9] compared to nonsyndromic patients (x˜ = 88.4[86.8-90.5], P ≤ .005.) CPAP was utilized following MDO in 2/24 (8.3%) of nonsyndromic and 16/31 (51.6%) of syndromic subjects (P ≤ .001,) for a median duration of 414 days. Three patients (5%) underwent tracheostomy, all had MDO. Nasogastric tube feeding at hospital discharge was more common following MDO (44, 80%) than CPAP-only (4, 44.4%, P ≤ .036), but did not differ at 6-month follow-up (P ≥ .376).ConclusionsCPAP appears to effectively reduce obstructive apnea in patients with RS and moderate TBAO and be a useful adjunct in syndromic patients following MDO with improved but persistent obstruction.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-30T11:23:19Z
      DOI: 10.1177/10556656221088173
       
  • Gender Disparities Among Craniofacial Surgeons

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      Authors: Brittany M Lala, Trina M Salvador, Fei Wang, Jinesh Shah, Joseph A Ricci
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo delineate career differences between genders of practicing plastic surgeons who have completed craniofacial fellowship given the known disparities in surgeons’ professional and personal lives and an already lengthy residency training, there is concern that less women may commit to further fellowship training.DesignCraniofacial fellowship programs were contacted to identify graduates and an internet search was attempted where information was not available. Surgeon profiles and literature databases were used to obtain practice demographics and publications.ParticipantsAccredited fellowships were identified through the American Society of Craniofacial Surgeons directory.ResultsProgram responses along with internet searches identified 201 graduates from 26 programs, of which 132 (66%) were men and 69 (34%) were women. On average, male graduates had 7.1 years in practice versus 6.6 years for females graduates (P = .176). There were significant differences between average number of publications (24.7 publications for males vs 14.1 for females, P = .009) and academic practice (46% males vs 64% females, P = .018). A similar percentage of males and females held leadership positions (13% males vs 16%, P = .552).ConclusionsDespite similar years in practice, men had significantly higher publications while women were significantly more likely to practice in an academic setting. Females are increasing their representation in academia and leadership within the craniofacial community. However, efforts must still be directed at increasing exposure to craniofacial surgery and supporting research and leadership pursuits earlier on during their careers.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-29T12:55:07Z
      DOI: 10.1177/10556656221089828
       
  • Exploration of Caregiver Interrater Agreement and Test-Retest Reliability
           on the Infant Cleft Observer Outcomes (iCOO)

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      Authors: Salene M. W. Jones, Todd C. Edwards, Brian G. Leroux, Kathleen A. Kapp-Simon, Donald L. Patrick, Laura P. Stueckle, Janine M. Rosenberg, Meredith Albert, Claudia Crilly Bellucci, Cassandra L. Aspinall, Katherine Vick, Carrie L. Heike
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundCaregiver and observer-reported measures are frequently used as outcomes for research on infants and young children who are unable to report on their own health. Our team developed the Infant with Clefts Observation Outcomes Instrument (iCOO) for infants with cleft lip with or without cleft palate. This exploratory study compared test-retest and interrater reliabilities to inform whether differences in caregiver perspective might affect the iCOO.MethodsThis study is a secondary analysis comparing caregiver interrater agreement to test-retest reliability. Twenty-five pairs of caregivers completed the iCOO before surgery, 1 week later for test-retest reliability, 2 days after surgery, and 2 months after surgery. Reliability was assessed using intraclass correlations (ICCs) and t-tests were used to compare ratings between caregivers.ResultsInfants had cleft lip (28%) or cleft lip and palate (72%). Primary caregivers were predominantly mothers (92%) and secondary caregivers were predominantly fathers (80%). Test-retest reliability met psychometric standards for most items on the iCOO (81%-86% of items). Caregiver agreement on the iCOO items was lower than test-retest reliability (33%-46% of items met psychometric standards). Caregivers did not systematically differ in whether they rated infants as healthier or less healthy than the other caregiver (5%-16% of items had statistically significant differences).ConclusionsCaregivers used the measure consistently, but had different experiences and perceptions of their infant’s health and functioning. Future studies are needed to explore mechanisms for the differences in test-retest and interrater reliability. Whenever possible, the same caregiver should provide ratings of the infant, including on the iCOO.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-28T07:43:36Z
      DOI: 10.1177/10556656221089155
       
  • SF3B4 Frameshift Variants Represented a More Severe Clinical Manifestation
           in Nager Syndrome

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      Authors: Zulvikar Syambani Ulhaq, Gita Vita Soraya, Lola Ayu Istifiani, Syafrizal Aji Pamungkas, William Ka Fai Tse
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Nager syndrome (NS) is a rare disease marked with craniofacial and preaxial limb anomalies. In this report, we summarized the current evidence to determine a possible genotype–phenotype association among NS individuals. Twenty-four articles comprising of 84 NS (including 9 patients with a severe form of NS [Rodriguez syndrome]) patients were examined, of which 76% were caused by variants in SF3B4 (OMIM *605593, Splicing Factor 3B, Subunit 4). Within the SF3B4 gene, variants located in exon 3 commonly occurred (20%) from a total identified variant, while hotspot location was identified in exon 1 (12%), and primarily occurred as frameshift variants (64%). Thirty-five distinct pathogenic variants within SF3B4 gene were identified with two common sites, c.1A > G and c.1060dupC in exons 1 and 5, respectively. Although no significant genotype–phenotype association was found, it is notable that patients with frameshift SF3B4 variants and predicted to lead to nonsense-mediated RNA decay (NMD) of the transcripts tended to have a more severe clinical manifestation. Additionally, patients harboring variants in exons 2 and 3 displayed a higher proportion of cardiac malformations. Taken together, this article summarizes the pathogenic variants observed in SF3B4 and provides a possible genotype–phenotype relationship in this disease.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-25T08:05:13Z
      DOI: 10.1177/10556656221089156
       
  • Primary Delayed Onset Craniosynostosis in a Child With ERF-Related
           Craniosynostosis Syndrome and Familial Cerebral Cavernous Malformation
           Syndrome

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      Authors: Stephanie Radu, Breanna Jedrzejewski, Leo Urbinelli
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Primary delayed onset craniosynostosis is defined as premature suture fusion that developed despite clear radiographic evidence of normal postnatal calvarial configuration and patent sutures earlier in life. It is rare in the literature and typically presents as secondary synostosis. In this brief clinical study, primary delayed onset craniosynostosis is described in its unique presentation at 4 years of age with a complex genetic history including ERF-related craniosynostosis syndrome and familial cerebral cavernous malformation syndrome. Although the delayed onset clinical course of ERF-related craniosynostosis syndrome has not been well described in the literature, our review suggests that it is distinctive to ERF-related craniosynostosis and should be considered when cases present without a history of trauma, when there is a positive family history, and particularly when cases present late onset; after 1 year of age.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-22T06:10:00Z
      DOI: 10.1177/10556656221088743
       
  • Supporting Our Cleft and Craniofacial Patients Beyond the Clinic:
           Scholarships to Promote Academic Achievement

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      Authors: Dillan F. Villavisanis, Nancy Folsom, Rachel A. McKenna, Jesse A. Taylor
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.

      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-21T08:04:01Z
      DOI: 10.1177/10556656221089154
       
  • What are the Soft Tissue Risk Factors for Obstructive Sleep Apnea in
           Patients with Down's Syndrome'

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      Authors: Dani Stanbouly, Barry Steinberg, Sung-Kiang Chuang
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo determine the risk factors and their respective magnitudes for developing Obstructive Sleep Apnea (OSA) in Down syndrome (DS) patients.DesignRetrospective cohort study.PatientsThe 2016 Kids’ Inpatient Database (KID) was queried to identify all patients diagnosed with DS.Main Outcome MeasuresThe primary predictor variables were tonsillar hypertrophy (TH), adenoidal hypertrophy (AH), Hypertrophy of Tonsils & Adenoids (HTA), Laryngeal Stenosis (LS), Hypotonia, Glossoptosis, Congenital Laryngomalacia (CL), and Overweight & Obesity (OO). The primary outcome variable was OSA.ResultsThe final sample consisted of 18,181 patients with a diagnosis of DS. Relative to patients aged 0-5, patients aged 6-10 (OR 3.5, P < 0.01), 11-5 (OR 3.4, P < 0.01), and 16 & above (OR 3.6, P < 0.01) were each independently associated with increased odds of OSA. Further, TH (OR 23.2, P < 0.01), AH (OR 20.3, P < 0.01), HTA (OR 64.2, P < 0.01), glossoptosis (OR 5.0, P < 0.01), CL (OR 4.3, P < 0.01), and OO (OR 3.7, P < 0.01) were all independent risk factors for OSA.ConclusionsThe presence of hypertrophied tonsils and adenoids together was the strongest risk factor for OSA. DS patients aged six and above were at risk for OSA development relative to younger patients. Patients with DS should be tested for OSA, which otherwise will deteriorate their existing comorbidities.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-21T08:03:21Z
      DOI: 10.1177/10556656221088171
       
  • Submucous Cleft Palate (SMCP): Indications and Outcomes of Radical Muscle
           Dissection Palatoplasty in Children Under 4 Years of Age

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      Authors: Ali M. Ghanem, Samim Ghorbanian, Tiffanie-Marie Borg, Debbie Sell, Brian C. Sommerlad
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo delineate the indications, referral sources and outcomes of radical muscle dissection palate repair in the first 4 years of life for patients with submucous cleft palate (SMCP).DesignThis study presents a retrospective analysis of a single surgeon's management.SettingAll children were treated at Great Ormond Street Hospital, United Kingdom.PatientsTwenty-three children with nonsyndromic SMCP were included in this study.InterventionsAll participants underwent radical muscle dissection repair before their fourth birthday.Outcome MeasuredParameters analyzed included: referral sources, indications for referral, extent of anatomical abnormality, and postoperative outcomes.ResultsPaediatricians provided the largest proportion of referrals to the cleft lip and palate clinic (39%) due to the presence of cleft lip/palate on prenatal scans or during neonatal examination. Over half (n = 12, 52%) of patients had severe anatomical anomalies being in grade IV (score 8-9), with the classical triad present to some extent in all but 2 of the patients. The main indication for surgery was nasal regurgitation of food and liquid (n = 9, 35%) followed by hypernasality (n = 6, 21%), difficulty feeding (n = 3, 8%), and severe anatomical defect (n = 2; 4%). Postoperatively, the presenting complaint improved in the majority of cases.ConclusionNonsyndromic SMCP is often missed, though may present with nasal regurgitation, feeding problems, and/or hypernasality, though may be missed in nonsyndromic children. Early radical muscle dissection repair in the first 4 years of life is safe and effective, facilitating normal speech development.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-21T08:02:25Z
      DOI: 10.1177/10556656221088170
       
  • Impact of Insurance Status on Initiation of Breast Milk Feeding Among
           Infants With CL ± P

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      Authors: Gabrielle Stowe, Eshan P. Schleif, Jamie L. Perry, Patrick M. Briley
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo evaluate rates of breast milk feeding among infants with cleft lip with or without cleft palate (CL ± P) enrolled in Medicaid compared to Private Insurance/Self-Pay.DesignThis was a population-based retrospective cohort study.SettingThe 2018 US National Vital Statistics System—Natality component (NVSS-N) was used to examine nationwide birth certificate data.ParticipantsInfants with cleft lip with or without cleft palate and either Medicaid or Private Insurance/Self-Pay were included.Main Outcome Measure:Breast milk feeding rates among infants with CL ± P, as a function of insurance status.ResultsChi-square tests of independence revealed that of 896 infants with CL ± P and insured by Medicaid, 527 (58.8%) were breast milk fed at discharge. Of 865 infants with CL ± P and insured by Private Insurance/Self-Pay, 621 (71.8%) were breast milk fed at discharge. Using logistic regression models and controlling for baseline demographic differences, results indicated that infants with CL ± P in the Medicaid group had reduced odds of breast milk feeding compared to the Private Insurance/Self-Pay group (OR = 0.08; 95% CI 0.56, 0.96).ConclusionBreast milk support services are often necessary for the initiation of breast milk feeding among infants with CL ± P. However, these resources are likely not as readily available for those enrolled in Medicaid. These results suggest that infants with CL ± P, enrolled in Medicaid, may experience reduced breast milk feeding rates due to limited resources to initiate breast milk feeding. Factors that may promote breast milk feeding among this population are discussed.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-21T08:01:46Z
      DOI: 10.1177/10556656221087553
       
  • 3D Photography to Quantify the Severity of Metopic Craniosynostosis

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      Authors: Madeleine K. Bruce, Wenzheng Tao, Justin Beiriger, Cameron Christensen, Miles J. Pfaff, Ross Whitaker, Jesse A. Goldstein
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aims to determine the utility of 3D photography for evaluating the severity of metopic craniosynostosis (MCS) using a validated, supervised machine learning (ML) algorithm.Design/Setting/PatientsThis single-center retrospective cohort study included patients who were evaluated at our tertiary care center for MCS from 2016 to 2020 and underwent both head CT and 3D photography within a 2-month period.Main Outcome MeasuresThe analysis method builds on our previously established ML algorithm for evaluating MCS severity using skull shape from CT scans. In this study, we regress the model to analyze 3D photographs and correlate the severity scores from both imaging modalities.Results14 patients met inclusion criteria, 64.3% male (n = 9). The mean age in years at 3D photography and CT imaging was 0.97 and 0.94, respectively. Ten patient images were obtained preoperatively, and 4 patients did not require surgery. The severity prediction of the ML algorithm correlates closely when comparing the 3D photographs to CT bone data (Spearman correlation coefficient [SCC] r = 0.75; Pearson correlation coefficient [PCC] r = 0.82).ConclusionThe results of this study show that 3D photography is a valid alternative to CT for evaluation of head shape in MCS. Its use will provide an objective, quantifiable means of assessing outcomes in a rigorous manner while decreasing radiation exposure in this patient population.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-21T08:01:13Z
      DOI: 10.1177/10556656221087071
       
  • Tessier's Cleft Number 6 Revisited: A Series of 26 new Cases and
           Literature Review of 44

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      Authors: Philippe Pellerin, Cristiano Tonello, Renato da Silva Freitas, Xiao Jun Tang, Nivaldo Alonso
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo fix a gray zone left in Tessier's classification of rare clefts with cleft 6 and to give a more comprehensive description of cleft 6 anatomy.DesignThe material used for the research was a series of 26 clinical cases of patients with assessed cleft 6 and 44 cases found out of a literature review with enough data to be useful. The 70 cases were cross-examined by the authors.Study SettingThe authors are senior craniofacial surgeons working in high-case load department from university centers where the patients are documented and receive primary as well as secondary treatment and follow-up.PatientsThe patients were selected out of the series of craniofacial deformities taken care of by the authors’ department as rare clefts.Main OutcomeWe describe the full spectrum of cleft 6 as an autonomous entity that could present itself in three subtypes: 6a is the most proximal and could be associated with cleft 8. The subtype 6b is medial toward the zygomatic arch and frequently associated with a bone and teeth appendage (frequently described as a “maxillary duplication”). The subtype 6C goes toward the external ear between the helix crus and the auditory meatus.ConclusionsThe Tessier's opinion is that Treacher Collins syndrome was the association of clefts 6, 7, and 8 and is no longer sustainable in the light of modern genetics. Most of the cleft 6 are misdiagnosed in the literature.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-14T11:36:59Z
      DOI: 10.1177/10556656221086459
       
  • Dental Anomalies in Parry-Romberg Syndrome: A 40-Year Retrospective Review

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      Authors: Ariana Aram, Alex Cappitelli, Fatma Dedeoglu, Ruth Ann Vleugels, Rich Bruun, Ingrid M. Ganske
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study investigates crown and root anomalies in patients with Parry-Romberg Syndrome.DesignThis is a retrospective review of patients with Parry-Romberg Syndrome who were evaluated at a tertiary care center from 1980-2020.SettingPatients seen in the dental unit from 1980-2020.Patients, ParticipantsSeventeen patients with documented Parry-Romberg Syndrome were referred for dental evaluation.Main Outcome MeasuresAll dental anomalies were documented. Root anomalies were assessed using panoramic radiographs and cone beam CT (CBCT) scans to evaluate buccal-lingual, mesio-distal, and axial measurements of hypoplastic teeth, which were compared to those of contralateral teeth.ResultsFindings included agenesis (29%, n = 5), hypoplastic teeth (29%, n = 5), delayed canine eruption (24%, n = 4), and mulberry molars (12%, n = 2). Of the five patients with tooth hypoplasia, four had CBCT records and the fifth had panoramic radiographs available for assessment. Axial length was always shorter in hypoplastic teeth relative to contralateral teeth, with differences ranging from 1.2-9.2 mm. Differences in crown size of hypoplastic versus contralateral teeth were unpredictable but always present.ConclusionsPatients with Parry-Romberg Syndrome can have hypoplastic roots with atypical crown morphology. A patient’s specific dental anomaly will influence planning and treatment.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-14T11:36:37Z
      DOI: 10.1177/10556656221086174
       
  • Three-dimensional Changes of Maxillary Alveolar Morphology After Using
           Modified Nasoalveolar Molding in Patients with Complete Unilateral Cleft
           lip and Palate

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      Authors: Nuntabhorn Chaisooktaksin, Jutharat Chimruang, Saran Worasakwutiphong, Ratchawan Tansalarak
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aimed to evaluate the three-dimensional changes in maxillary alveolar morphology after using modified NAM in patients with complete unilateral cleft lip and palate.DesignThis is a retrospective study.SettingThe study was carried out in the dental hospital, Faculty of Dentistry of Naresuan University, Phitsanulok, which serves as a tertiary care center.PatientsThe population sample consisted of 19 patients with nonsyndromic complete unilateral cleft lip and palate.InterventionAll patients received the modified NAM treatment based on the treatment protocol of the Naresuan University Cleft and Craniofacial Center, Thailand.Main Outcome MeasureDental models obtained at pre-treatment (T0) and post-treatment (T1) were scanned to construct the digital models. The maxillary digital models that showed dimensional changes between T0 and T1 were measured using a computer graphic software.ResultsThe modified NAM resulted in a significant decrease in the anterior cleft width, posterior cleft width, and anterior arch width. Conversely, it caused a significant increase in the length of the lesser cleft segment and the greater segment rotation. However, the change in the height of both segments and posterior arch width was not found to be significant.ConclusionsThe modified NAM was an effective device for reducing the alveolar cleft width while improving the alignment of alveolar cleft segments.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-14T01:32:59Z
      DOI: 10.1177/10556656221086816
       
  • Wound Healing Following Palatoplasty Using Either Honey or Warm Saline
           Mouth Bath for Postoperative Wound Care: A Randomized-Controlled Study

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      Authors: Adegbayi Adeola Adekunle, Olutayo James, Emmanuel Onyebuchi Onuoha, Wasiu Lanre Adeyemo
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo evaluate and compare the effect of honey or saline mouth bath for wound care on the rate of re-epithelisation of the lateral palatal defects and occurrence of early postoperative complications following palatoplasty.MethodologyThis was a prospective randomized study on participants with non-syndromic cleft palate conducted at a tertiary health institution in Lagos, Nigeria. The test group received oral honey drops for post-operative care for an initial period of two weeks post-surgery, while the control group had regular oral toileting using a warm saline solution. The primary outcome was epithelisation of lateral palatal defect at 2- and 4-weeks post repair. Descriptive and comparative statistics were computed, and the p-value was set at
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-09T10:11:43Z
      DOI: 10.1177/10556656221086192
       
  • An Algorithmic Approach for Deploying Buccal Fat Pad Flaps and Buccal
           Myomucosal Flaps Strategically in Primary and Secondary Palatoplasty

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      Authors: Fatima Qamar, Mariel M. McLaughlin, Matthew Lee, Aleshia J. Pringle, Jordan Halsey, S. Alex Rottgers
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveRecent publications have introduced the use of buccal myomucosal and fat pad flaps to augment palatal repairs with autologous tissue. We propose a workflow for intraoperative decision-making to introduce these adjuncts into standard palatoplasty procedures.Design/PatientsA retrospective chart review of a single-surgeon series of patients undergoing primary and secondary palatoplasties performed between October 2017 and November 2020 was completed after Institutional Review Board approval.Main Outcome MeasuresPatient demographics, phenotype, operative details, and postoperative complications were recorded.ResultsFifty-eight patients were included in a review. For those undergoing primary repair, 23.3% underwent a Furlow palatoplasty alone, 46.3% had a Furlow palatoplasty accompanied with acellular dermal matrix (ADM) and/or a buccal fat flap (BFF). A unilateral buccal myomucosal flap (BMMF) with or without augmentation with BFF or ADM was employed in 16.3% of the cases. Fourteen percent required a bilateral BMMF+/− ADM. Fistula occurrence was 2.3% (n = 1). For revisions, 27% underwent only a conversion Furlow palatoplasty, 26% had a conversion Furlow palatoplasty accompanied with ADM and/or a BFF, 33% had a unilateral BMMF or BMMF/ADM, and 14% required a bilateral BMMF+/− ADM.ConclusionsIn severe phenotypes or complicated cases, buccal fat flaps and myomucosal flaps may be utilized. This approach has mostly replaced the use of ADM over time. An algorithmic approach to palatoplasty allows surgeons to tailor the extent of surgery to the needs of each patient.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-09T04:05:16Z
      DOI: 10.1177/10556656221084879
       
  • Open Versus Minimally Invasive Approach for Craniosynostosis: Analysis of
           the National Surgical Quality Improvement Program-Pediatric

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      Authors: Imran Rizvi, Lucas M. Harrison, Shyon Parsa, Rami R. Hallac, James R. Seaward, Alex A. Kane
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis multicenter study aimed to compare demographic, operative, and short-term outcomes data between open and minimally invasive surgical approaches for craniosynostosis repair utilizing the American College of Surgeon’s National Surgical Quality Improvement Program Pediatric (NSQIP-P) database and highlight surgical disparities among races and ethnicities.DesignRetrospective review of large multicenter database.SettingFreestanding general acute care children's hospitals, children's hospitals within a larger hospital, specialty children's hospitals, or general acute care hospitals with a pediatric wing.Patients and ParticipantsA total of 4931 pediatric patients underwent craniosynostosis correction within the NSQIP-P database from 2013 to 2019.InterventionsNone.Main Outcome Measure(s)Demographic information included age at surgery, sex, race, and ethnicity. Operative and outcomes measures included operative time, anesthesia time, days until discharge, postoperative complications, blood transfusions, 30-day readmission, and 30-day unplanned return to operating room.ResultsPatients who underwent minimally invasive surgery had significantly shorter operative and anesthesia times (p 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-07T09:56:15Z
      DOI: 10.1177/10556656221085478
       
  • The Impact of Fan-Type Rapid Palatal Expanders on Speech in Patients With
           Unilateral Cleft Lip and Palate

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      Authors: Virginia Bertucci, Kyle Stevens, Nicole Sidhu, Sunjay Suri, Tim Bressmann
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivesRapid palatal expanders (RPEs) are commonly used in patients with cleft lip and palate (CLP) prior to secondary alveolar bone grafting (SABG). Their position and size can impede tongue movement and affect speech. This study assessed changes in perception and production of speech over the course of RPE treatment.DesignProspective longitudinal.SettingTertiary university-affiliated hospital.ParticipantsTwenty-five patients with unilateral CLP treated with Fan-type RPEs, and their parents.InterventionsPatient and parent speech questionnaires and patient speech recordings were collected at baseline before RPE insertion (T1), directly after RPE insertion (T2), during RPE expansion (T3), during RPE retention (T4), directly after RPE removal but before SABG (T5), and at short-term follow-up after RPE removal and SABG (T6).Main Outcome MeasuresRatings for patient and parent questionnaires, first (F1) and second (F2) formants for vowels /a/, /i/, and /u/, and nasalance scores for non-nasal and nasal sentences, were obtained and analyzed using mixed model analyses of variance.ResultsRatings worsened at T2. For the vowel /a/, F1 and F2 were unchanged at T2. For the vowel /i/, F1 increased and F2 decreased at T2. For the vowel /u/, F1 was unchanged and F2 decreased at T2. Nasalance was unchanged at T2. All outcome measures returned to T1 levels by T4.ConclusionsRPE insertion resulted in initial adverse effects on speech perception and production, which decreased to baseline prior to removal. Information regarding transient speech dysfunction and distress may help prepare patients for treatment.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-07T09:56:10Z
      DOI: 10.1177/10556656221084541
       
  • Childhood Experiences and Perspectives of Individuals With Orofacial
           Clefts: A Qualitative Systematic Review

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      Authors: Emilija D. Jensen, Brianna F. Poirier, Kelly J. Oliver, Rachel Roberts, Peter J. Anderson, Lisa M. Jamieson
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveChildren and adolescents with orofacial clefts may experience ongoing psychosocial impacts due to the continuous nature of cleft treatments, facial and dental differences, and speech and hearing difficulties. The aim of this qualitative systematic review was to better understand the experiences of children and adolescents with orofacial clefts.DesignA systematic search strategy using PubMed, Embase, Emcare, Scopus, and Web of Science databases was performed to identify relevant qualitative studies evaluating the lived experience of children and adolescents with orofacial clefts from inception through to June 2021. Eligible studies were critically appraised using the Joanna Briggs methodology and a meta-aggregative approach.ResultsThe search identified 2466 studies, with 13 found to meet the inclusion criteria. Extraction of 155 findings resulted in 27 categories, which were meta-aggregated into 7 overarching synthesized findings. These 7 core findings included aspects of child experience and findings that enhanced or impeded child experience at the individual, family, and community levels.ConclusionsFactors that impeded child experience at the individual, family, and community levels were more pronounced than factors that enhanced their experience among children and adolescents with orofacial clefts. Further initiatives are needed to provide support to individuals, families, and school communities to enhance children's experience of orofacial cleft during the formative childhood and adolescent years.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-07T04:05:19Z
      DOI: 10.1177/10556656221084542
       
  • Triple Layer Oronasal Fistula Repair Using Local Endonasal Flaps: Case
           Series of Three Patients

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      Authors: Joseph N. Badaoui, Garret Choby, Ryan A. McMillan, Andrew J. Goates, Shelagh A. Cofer
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Oronasal fistula (ONF) is a common complication encountered after palatoplasty. Repair is indicated when symptoms impact speech and swallowing. In spite of the variety of surgical approaches described to repair these defects, recurrence rates remain high. Traditionally, successful closure is said to be achieved in using a double-layered approach due to the three-dimensional aspect of the defect. The extent of the fistula into the nasal cavity has incited an increased curiosity in using local endonasal flaps. In recent years, endonasal reconstructive procedures have seen increased interest and application, from cranial base defect repairs to orbital reconstruction and beyond. The nasoseptal (NSF) and inferior turbinate flaps (ITF) possess a robust arterial supply and an exceptional reach with excellent results demonstrated in large defect repair. However, the use of these flaps in ONF repair is scarcely discussed in the literature, and their effectiveness is relatively undetermined. In this manuscript, we present a series of three patients who underwent a triple layer ONF closure, with the oral portion incorporating a turn-in mucosal flap plus a local palate rotation flap or greater palatine artery pedicled-rotation flap, and a NSF or an ITF for the nasal portion of the defect.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-04T02:26:40Z
      DOI: 10.1177/10556656211062040
       
  • Three-dimensional Analysis of Factors Related to the Effective Alveolar
           Molding in Presurgical Infant Orthopedics: Findings From a Pilot Study

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      Authors: Mai Fujimoto, Mikiko Mano, Au Sasaki, Hiroya Hasegawa, Masaharu Suzuki, Naoko Nojiri, Yuri Minami, Kazuyuki Tokioka, Naoto Suda
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivePresurgical infant orthopedics (PIO) reduces the severity of the original cleft and burden on patients and their parents, provides better esthetics and function, and enables surgeons to achieve better surgical repair. To reduce the alveolar cleft width and to predict treatment difficulty using PIO, various measures were examined in pretreatment cast models.DesignRetrospective case-control pilot study.PatientsThe patients were 22 infants with non-syndromic unilateral cleft lip and palate (UCLP), and cast models of these infants were used.MethodsAfter PIO using passive plates, infants with UCLP were divided into two groups: contact group (12 cases with close proximity of the greater and lesser segments) and non-contact group (10 cases without proximity of segments). The two groups were compared, and variables related to the proximity between alveolar clefts were examined.ResultsThere was no significant difference in age at PIO initiation between the two groups. However, the treatment duration was significantly longer in the non-contact group than in the contact group. Among the 13 variables, the initial lateral deviation of the nasal septum was significantly larger in the contact group than in the non-contact group. A significant positive correlation was observed between the initial lateral deviation of the nasal septum and reduction of the alveolar cleft width by PIO.ConclusionInitial lateral deviation of the nasal septum is a predictive factor for the proximity between alveolar segments in infants with UCLP at the PIO.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-02T12:26:37Z
      DOI: 10.1177/10556656221083804
       
  • A Novel and Practical Protocol for Three-Dimensional Assessment of
           Alveolar Cleft Grafting Procedures

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      Authors: Celine C. Stoop, Nard G. Janssen, Timen C. ten Harkel, Antoine J. W. P. Rosenberg
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo evaluate the reproducibility and accuracy of a new, easy-to-use volumetric assessment of the alveolar cleft.DesignTwelve cone-beam computed tomography (CBCT) datasets of patients with a unilateral cleft lip, alveolus, and palate were evaluated by two investigators. Residual alveolar cleft calcified volume one year after surgery was analyzed by using standardized landmarks to determine the borders of the cleft defect and semi-automatically segment the alveolar cleft defect.ResultsThe Dice-coefficient between observers for the segmented preoperative alveolar cleft defect was 0.81. Average percentage of residual alveolar cleft calcified material was 66.7% one year postoperatively.ConclusionsThis study demonstrates a reliable and practical semi-automatic three-dimensional volumetric assessment method for unilateral clefts using CBCT.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-02T12:26:14Z
      DOI: 10.1177/10556656221074210
       
  • Application of a Novel Nasal Clip for Nostril Retention After Primary
           Unilateral Cleft Rhinoplasty

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      Authors: Shiming Zhang, Chao Yang, Min Wu, Jiayi Yin, Ni Zeng, Bing Shi, Hanyao Huang
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study aimed to investigate the outcomes of using a novel nasal clip designed for nostril retention after primary unilateral cleft rhinoplasty.DesignThis is a retrospective study.SettingDepartment of Cleft Lip and Palate in a stomatological hospital.PatientsA retrospective study was conducted on 57 patients who had a unilateral complete cleft lip without cleft palate and underwent primary surgical repair.InterventionsThe patients were categorized into 3 groups: the group in which the conventional nasal retainer was used, that in which the nasal clip was used, and the control group in which no postoperative nasal retainer was used. The nasal retainer or our nasal clip was applied 7 days after primary surgical repair and kept in the nostrils of children from 6 to 12 months.Main Outcome MeasuresNoses underwent photogrammetry preoperatively, postoperatively, and at follow-up. The nose was also subjectively scored at the follow-up.ResultsThe nasal retainer and the nasal clip significantly minimized relapse as determined by nasal tip deviation. The nasal clip sustained nasal symmetry, as determined by the postoperative nostril width ratio, better than did the conventional retainer. After 6 to 12 months, the nasal clip achieved better columellar morphology and a more symmetric nasal base than did the conventional nasal retainer. Statistical differences in subjective scores between the control and nostril retention groups were noted.ConclusionThe application of the novel nasal clip after primary unilateral cleft rhinoplasty preserves nasal morphology and reduces postoperative relapse without the need for adhesive tape.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-02T04:17:32Z
      DOI: 10.1177/10556656221083812
       
  • A Single Stage Composite Cleft Septorhinoplasty for Correction of the
           Mature Unilateral Cleft Nose Deformity - The Gujrat Technique

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      Authors: Diaa Othman, Mark Gorman, Muhammad Adil Abbas Khan, Yangmyung Ma, Dujanah Siddique Bhatti, Sadia Rafiq, Hussan Berkhez Shami, George Lye, Michael McBride, Muhammad Riaz
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo evaluate the results of a single stage composite cleft septorhinoplasty procedure (“The Gujrat Technique”) to correct the exaggerated cleft nose deformity after completion of nasal growth in an adult patient cohort.MethodsAdult patients with a residual unilateral cleft nasal deformity were deemed eligible for the proposed “Gujrat Technique”. Over a 10-year period (2007–2017), 96 adult patients underwent this composite cleft septorhinoplasty as a single stage operation. Post-operative nasal symmetry evaluation was undertaken using the validated computer program ‘SymNose’. Functional outcome and patient satisfaction were assessed using Nasal Obstruction Symptom Evaluation scale and Rhinoplasty Outcome Evaluation (ROE) questionnaires respectively. Various statistical analysis methods were used to validate the obtained results.ResultsDue to poor compliance with follow-up, post-operative assessments were undertaken in only 32 patients. The single group study design using the non-parametric matching pairs Wilcoxon Sign test (p 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-03-02T04:17:23Z
      DOI: 10.1177/10556656221082765
       
  • Clinical Application of Hyperdry Amniotic Membrane in Cleft Palate Repair

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      Authors: Kumiko Fujiwara, Hiroaki Tsuno, Motonori Okabe, Toshiko Yoshida, Shuichi Imaue, Kei Tomihara, Naoya Arai, Makoto Noguchi
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo examine the safety and efficacy of hyperdry amniotic membrane (HDAM) for wound closure after palatoplasty in cleft palate patients.MethodsHDAMs were prepared by washing and drying under infrared rays and microwaves at temperatures less than 60°C using a hyperdrying device. A total of 16 cleft palate patients (8 males, 8 females), aged 1 to 3 years (mean age 1 year 9 months), received one-stage pushback palatoplasty. The remaining raw wound after surgery was covered by an HDAM and a plastic cover plate. The cover plate was removed 1 week after surgery and parameters including temperature, feeding, allergic reactions, postoperative bleeding, re-epithelialization, wound dehiscence, and infection were monitored during the follow-up period of 31.2 months.ResultsAll patients could adequately ingest at 5 days postoperation and after removal of the cover plate. None of the patients had a persistent fever or allergic reactions. Ingestion was feasible immediately in all patients, and no postoperative bleeding was observed during ingestion. No secondary hemorrhages were observed during follow-up. No postoperative wound dehiscence on the midline of the palate was observed. No infections were observed after the removal of the cover plate. No patients suffered from severe scar formation or contracture of the wound in the follow-up period. Hemorrhage, undue epithelialization, and scar contracture did not occur in any patient. The mean evaluation score was 7.75 points.ConclusionHDAM can be used safely and effectively for wound closure following palatoplasty in cleft palate infants. Future studies testing the safety of patient's own amnion for palatoplasty, are required.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-28T11:55:50Z
      DOI: 10.1177/10556656221075937
       
  • Variability and Excess in Opioid Prescribing Patterns After Cleft and
           Craniosynostosis Repairs

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      Authors: Ema Zubovic, Gary B. Skolnick, Jacob D. AuBuchon, Erika A. Waters, Alison K. Snyder-Warwick, Kamlesh B. Patel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo critically analyze pediatric opioid prescription patterns after cleft and craniosynostosis repairs.DesignObservational study 1) retrospectively reviewing pediatric opioid prescriptions from July 2018 to June 2019 and 2) prospectively surveying patients about actual opioid use from August 2019 to February 2020.SettingAcademic tertiary care pediatric hospital.Patients133 pediatric patients undergoing cleft lip and/or palate or craniosynostosis repairs. Prospective surveys were offered at postoperative visits; 45 of 69 eligible patients were enrolled.InterventionNone.Main Outcome MeasuresOpioid doses prescribed at discharge and actual home opioid use.Results90 patients with cleft lip and/or palate and 43 patients with craniosynostosis were included. Median prescribed opioid doses were 10.3 for cleft lip and/or palate procedures (range 0–75), and 14.3 for craniosynostosis repairs (range 0–50). In patients with cleft lip and/or palate, there was a negative correlation between age at surgery and prescribed opioid doses (rs  =  -0.228, p  =  0.031). 45 patients completed surveys of home opioid use. No patients used more than 10 doses. Forty percent used no opioids at home, 33% used 1 to 2 doses, 18% used 3 to 5 doses, and 9% used 6 to 10 doses.ConclusionsOpioid prescriptions vary widely after common craniofacial procedures. Younger patients with cleft lip and/or palate may be more likely to be prescribed more doses. Actual home opioid use is less than prescribed amounts, with most patients using five or fewer doses. A prescribing guideline is proposed.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-28T03:32:17Z
      DOI: 10.1177/10556656221083082
       
  • Epidemiology of Nonsyndromic, Orofacial Clefts in Texas: Differences by
           Cleft Type and Presence of Additional Defects

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      Authors: Maria Luisa Navarro Sanchez, Michael D. Swartz, Peter H. Langlois, Mark A. Canfield, A.J. Agopian
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo describe the current epidemiology of nonsyndromic cleft palate alone (CP) and cleft lip with or without cleft palate (CL ± P) in Texas and examine differences in the characteristics of infants with CP and CL ± P based on the presence/absence of additional defects.DesignWe used data from the Texas Birth Defects Registry, a statewide active birth defect surveillance system, from 1815 cases with CP and 5066 with CL ± P, without a syndrome diagnosis (1999-2014 deliveries). All live births in Texas were used for comparison. Poisson regression was used to calculate crude and adjusted prevalence ratios (aPR) for each characteristic, separately for each cleft subphenotype.ResultsThe prevalence of CL ± P and CP in our study was estimated as 8.3 and 3.0 per 10 000 live births, respectively. After adjusting for several characteristics, several factors were associated with CL ± P, CP, or both, including infant sex and maternal race/ethnicity, age, smoking, and diabetes. There were several differences between infants with isolated versus nonisolated clefts. For example, maternal prepregnancy diabetes was associated with an increased prevalence of CL ± P (aPR 7.91, 95% confidence interval [CI]: 5.53, 11.30) and CP (aPR 3.24, 95% CI: 1.43, 7.36), but only when additional defects were present.ConclusionsFindings from this study provide a contemporary description of the distribution of orofacial clefts in Texas accounting for differences between isolated and nonisolated clefts. They may contribute to increasing our understanding of the etiology of CP and CL ± P.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-28T03:31:59Z
      DOI: 10.1177/10556656221080932
       
  • Current Surgical Practice for Children Born with a Cleft lip and/or Palate
           in the United Kingdom

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      Authors: Matthew Fell, Alex Davies, Amy Davies, Shaheel Chummun, Alistair R.M. Cobb, Kanwalraj Moar, Yvonne Wren
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study describes primary surgical reconstructions performed for children born with a cleft lip and/or palate (CL ± P) in the United Kingdom (UK).DesignData forms completed at the time of surgery included details on timing, technique, and adjuncts used during the operative period. Demographic data on participants were validated via parental questionnaires.SettingData were obtained from the Cleft Collective, a national longitudinal cohort study.PatientsBetween 2015 and 2021, 1782 Cleft Collective surgical forms were included, relating to the primary reconstructions of 1514 individual children.ResultsThe median age at primary cheiloplasty was 4.3 months. Unilateral cleft lips (UCL) were reconstructed with an anatomical subunit approximation technique in 53%, whereas bilateral cleft lips (BCL) were reconstructed with a broader range of eponymous techniques. Clefts of the soft palate were reconstructed at a median age of 10.3 months with an intravelar veloplasty in 94% cases. Clefts of the hard palate were reconstructed with a vomer flap in 84% cases in a bimodal age distribution, relating to reconstruction carried out simultaneously with either lip or soft palate reconstruction. Antibiotics were used in 96% of cases, with an at-induction-only regimen used more commonly for cheiloplasties (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-24T11:36:26Z
      DOI: 10.1177/10556656221078151
       
  • Delayed Presentation of Unicoronal Craniosynostosis—Masked by
           Ipsilateral Posterior Deformational Plagiocephaly

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      Authors: Ian Chow, Alyssa K. Brisbin, Erin E. Anstadt, Jesse A. Goldstein, Joseph E. Losee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Despite a greater awareness of the differential diagnosis of head shape abnormalities among pediatricians, the effect of deformational forces on calvarial morphology can complicate the diagnosis of craniosynostosis. In this report, we describe 2 patients diagnosed with unicoronal craniosynostosis (UCS) in a delayed fashion due to the presence of concomitant posterior deformational plagiocephaly (PDP). In both cases, the severity of each patients’ PDP obscured changes typically associated with UCS. This unique presentation underscores the importance of having a high index of suspicion for possible premature suture fusion despite the presence of concomitant PDP.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-23T01:38:11Z
      DOI: 10.1177/10556656221080996
       
  • The Impact of the Covid-19 Pandemic on Cleft Lip and Palate Service
           Delivery for New Families in the United Kingdom: Medical and Community
           Service Provider Perspectives

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      Authors: Danielle McWilliams, Bruna Costa, Sabrina Blighe, Marc C. Swan, Matthew Hotton, Nichola Hudson, Nicola Marie Stock
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivesProfessionals in the United Kingdom providing care to new families affected by cleft lip and/or palate (CL/P) had to adapt to ensure families’ needs were met during a time of uncertainty due to Covid-19. The aims of this study were to explore the impacts of the pandemic on CL/P care provision for new families from the perspectives of professionals working in medical and community settings along with any personal impact on professionals and their reflections on the future of CL/P care.DesignSemistructured interviews (n  =  27) were completed about experiences from March 2020 to October 2020 with consultant cleft surgeons (n  =  15), lead clinical nurse specialists (n  =  8), and staff working at the Cleft Lip and Palate Association (n  =  4). Transcripts were analyzed using inductive thematic analysis.ResultsThree themes were identified: (1) the impact of Covid-19 on the provision of cleft care in the United Kingdom, including working conditions, delays to treatment, and Covid-19 policies; (2) the impact of the pandemic on professionals’ mental health, including personal distress and concerns about Covid-19 exposure; and (3) reflections on the future of CL/P care, whereby professionals expressed both hope and concern about the Covid-19 recovery effort.ConclusionsThe ongoing Covid-19 pandemic has impacted CL/P service delivery for new families significantly, warranting recommendations for cohesive psychological support for families in addition to a safe and resourced recovery effort. Support for professionals is also suggested, following existing evidence-based models for providers’ needs that address the difficulties of working throughout challenging times.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-23T01:37:40Z
      DOI: 10.1177/10556656221074870
       
  • Cone-Beam Computed Tomographic Assessment of Maxillary Sinus
           Characteristics in Patients With Cleft Lip and Palate: A Systematic Review
           and Meta-Analysis

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      Authors: Sukeshana Srivastav, Nitesh Tewari, Ritu Duggal, Shubhi Goel, Morankar Rahul, Vijay Prakash Mathur, Rahul Yadav, Ashish Dutt Upadhyaya
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe aim of this systematic review and meta-analysis was to evaluate the maxillary sinus characteristics of patients with cleft lip and palate (CLP).DesignThe study included manuscripts which met the following criteria: (1) study of individuals with CLP in any age group or gender and (2) study of individuals in whom assessment of maxillary sinus characteristics had been done by cone-beam computed tomography. Studies with (1) individuals having special health-care needs and (2) individuals with any syndrome affecting the development of the head and neck were excluded.ResultsIn total, 11 articles were included in the review, based on the inclusion and exclusion criteria. Pooled maxillary sinus volume (MSV) on the cleft sides of patients with unilateral cleft lip and palate (UCLP) was found to be 9433.14 mm3 (95% CI, 7453.99-11 412.30), which was significantly smaller than that of controls. The meta-analyses also revealed significantly reduced MSV on the cleft sides of patients with UCLP. The differences between cleft and noncleft sides of the patients with UCLP were not found to be statistically significant. Strength-of-evidence was found to be moderate in 4 characteristics, along with 10 out of 11 articles showing low risk-of-bias.ConclusionsIt was concluded that MSV was reduced and mucosal thickening was increased/present in patients with CLP as compared with controls. The differences between MSV of cleft and noncleft sides of the patients with UCLP could not be established. However, heterogeneity was found in terms of sample size, stratification of samples by age, and evaluation of some confounding factors.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-18T03:33:59Z
      DOI: 10.1177/10556656221080359
       
  • Population Prevalence and Trends of Oral Clefts in Colombia: Analysis by
           Departments

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      Authors: Herney Alonso Rengifo Reina, Guarnizo Peralta Stefany Brigetty, Yezioro Rubinsky Salomón
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Objective:Determine the population prevalence and trends of cleft lip and/or palate (CL/P) by department for Colombia in the period 2009 to 2015.Methods:Prevalence study based on Individual Registry of Health Services in general population from 2009 to 2015. All people diagnosed with CL/P were included for all ages, type of diagnosis, and any type of health services in the mentioned period. The prevalence was calculated by period and point for each year, for each department and according to the type of cleft. Stationarity on time series was evaluated using (Dickey-Fuller) and (Phillips-Perron). A trend test was applied to estimate whether the increase in prevalence was significant in the period. The trend test used was a Poisson regression.Results:A total 15 225 people with CL/P were identified, where 53.3% were men. The national period prevalence of CL/P is 3.37 per 10 000 with upward trend (prevalence ratio = 1.34, P = .05) and nonstationary behavior. The national period prevalence of cleft lip is 0.93 per 10 000, cleft palate 1.17 per 10 000, and cleft lip and palate (CLP) 1.26 per 10 000, where CLP is subclassify into unilateral CLP (0.83 per 10 000) and bilateral CLP (0.43 per 10 000). At the departmental level, the highest CL/P prevalence is Guaviare (11.2), followed by Guainía (8.4) and the lowest Quindío (0.49).Conclusions:In Colombia, the national period prevalence of CL/P is 3.37 per 10 000 with upward trend at national level indicates an increase in prevalence from 2009 to 2015. The 77% of the total CL/P population are infant or adolescent. Geographically, the central region has the highest availability of technologies.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-18T03:33:42Z
      DOI: 10.1177/10556656221078148
       
  • Quality Assurance Standards for Outreach Cleft Lip and Cleft Palate Repair
           Programs in Low-Resource Settings

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      Authors: Elsa M. Chahine, Elie P. Ramly, Alexander P. Marston, Sarena N. Teng, Marie Nader, Serena N. Kassam, Rami S. Kantar, Usama S. Hamdan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundComprehensive cleft care is a multidisciplinary team endeavor. While untreated craniofacial conditions have multiple undue repercussions, cleft care in outreach settings can be fraught with significant perioperative morbidity risks.AimPropose updated quality assurance standards addressing logistic and operational considerations essential for the delivery of safe and effective cleft lip and /or palate (CL/P) care in low and middle-income countries (LMICs) settings.MethodsBased on American Cleft Palate-Craniofacial Association (ACPA) quality standards, published literature, published protocols by Global Smile Foundation (GSF), and the senior author's three-decade experience, updated standards for outreach cleft care were synthesized.ResultsTen axes for safe, effective, and sustainable cleft lip and palate care delivery in underserved settings were generated: 1) site assessment, 2) establishment of community partnerships, 3) team composition and credentialing, 4) team training and mission preparation, 5) implementation of quality assurance guidelines, operative safety checklists, and emergency response protocols, 6) immediate and long-term postoperative care, 7) medical record keeping, 8) outcomes evaluation, 9) education, and 10) capacity building and sustainability. Subsequent analysis further characterized essential components of each of those ten axes to delineate experience derived and evidence-based recommendations.DiscussionQuality assurance guidelines are essential for the safe delivery of comprehensive cleft care to patients with CL/P in any setting. Properly designed surgical outreach programs relying on honest community partnerships can be effectively used as vehicles for local capacity building and the establishment of sustainable cleft care ecosystems.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-18T03:33:35Z
      DOI: 10.1177/10556656221074883
       
  • Early Secondary Alveolar Bone Grafting and Facial Growth of Patients with
           Complete Unilateral Cleft Lip and Palate

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      Authors: Jean-Charles Doucet, Kathleen A. Russell, John Daskalogiannakis, Ana M. Mercado, Ronald R. Hathaway, Gunvor Semb, William C. Shaw, Ross E. Long
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo investigate the craniofacial growth outcomes of early secondary alveolar bone grafting(ABG) around 6 years of age.DesignRetrospective cohort studySetting1 North-American and 5 Northern-European cleft centers.Subjects33 subjects with CUCLP consecutively treated with secondary ABG around 6 years of age were compared to 105 subjects from 4 centers treated with late secondary ABG and 19 subjects from 1 center with primary ABG.MethodsPreorthodontic standardized lateral cephalometric radiographs taken after 12 years of age were traced and analyzed according to the Eurocleft Study protocol. Fourteen angular and two proportional measurements were performed. Measurement means from the Study Center(SC) were compared to 5 Northern-European centers using analysis of variance and Welch's modified t-tests, and P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-16T02:19:59Z
      DOI: 10.1177/10556656221080990
       
  • The Public Health Importance of Flaviviruses as an Etiological
           Environmental Factor in Nonsyndromic Cleft Lip and/or Palate: In silico
           Study

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      Authors: Kaique Cesar de Paula Silva, Thiago Silva Messias, Simone Soares
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis in silico study aims to investigate flaviviruses as an environmental factor in the etiology of nonsyndromic cleft lip and/or palate (CL/P).DesignA scoring method with 7 topics—disease, transplacental passage, tropism, cellular damage, reported case, analysis of genome similarity, and transcriptome between virus and host, was created based on literature and in silico experimentation. Viral genomes of NCBI virus were obtained and BLAST 2.12.0 was applied for the similarity analysis, adjusted to search for only human sequences related to CL/P with the statistical threshold defined for E-value ≤1.ResultsFlaviviruses with high potential to cause CL/P were: serotypes 2, 3, and 4 of the Dengue virus and lineage 2 of the West Nile virus, while the Yellow Fever virus, Japanese encephalitis virus, Tick-borne encephalitis virus, and Saint Louis encephalitis virus presented with medium potential to cause CL/P. As for the Zika virus, even strains associated with microcephaly showed only medium potential.ConclusionsDengue virus and West Nile virus presented with high potential to act as environmental factors in the etiology of CL/P.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-15T09:14:18Z
      DOI: 10.1177/10556656221074206
       
  • Timing and Duration of Facial Nerve Dysfunction After Mandibular
           Distraction Osteogenesis for Robin Sequence

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      Authors: Hannah R. Crowder, Esperanza Mantilla-Rivas, Elina Kapoor, Monica Manrique, Jason Stein, Jacob S. Nasser, Taeun Chang, Gary F. Rogers, Albert K. Oh
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveCollect data from craniofacial surgeons to analyze mandibular distraction osteogenesis (MDO) protocols, and facial nerve dysfunction (FND) to characterize this common, but poorly documented complication after MDO in infants with Robin Sequence (RS).Design, Setting, and ParticipantsA 16-question anonymous survey designed through REDCap was digitally distributed to members of the American Cleft Palate-Craniofacial Association and International Society of Craniofacial Surgery (ISCFS).Main Outcome Measure(s)Demographic information, MDO perioperative variables, surgeon experience with FND after MDO for patients with RS, and the timing and duration of FND were analyzed.ResultsEighty-four responses were collected, with 80 included for analysis. Almost two-thirds of respondent surgeons reported FND as a complication of MDO in patients with RS (51, 63.8%); 58.8% (n   =   47) transient FND and 5% (n   =   4) with permanent facial nerve palsy only. Both transient and permanent FND was documented by 13 (16.3%) respondents. Among respondents, FND was observed immediately following initial device placement/osteotomies in 45.1%, during distraction in 45.1%, during consolidation in 19.6%, and following device removal in 43.1%. Twenty-five of these respondent surgeons reported resolution of FND between 1 and 3 months (53.2%, n   =   25).ConclusionsFND after MDO in patients with RS was noted by most respondents in this survey study. While most surgeons noted temporary FND, one-fifth reported long-term dysfunction. FND was documented most commonly following device placement/osteotomies or during active distraction. Further research should seek to establish risk factors associated with FND and identify surgical and perioperative prevention strategies
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-15T05:35:44Z
      DOI: 10.1177/10556656221077591
       
  • Enhanced Recovery After Cleft Lip Repair: Protocol Development and
           Implementation in Outreach Settings

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      Authors: Antonio M. Melhem, Elie P. Ramly, Omar S. Al Abyad, Elsa M. Chahine, Sarena Teng, Raj M. Vyas, Usama S. Hamdan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      IntroductionClefts of the lip are of the most common congenital craniofacial anomalies. The development and implementation of an enhanced recovery after surgery (ERAS) protocol among patients undergoing cleft lip repair may decrease postoperative complications, accelerate recovery, and result in earlier postoperative discharge.MethodsA modified ERAS program was developed and applied through Global Smile Foundation outreach craniofacial programs. The main components of this protocol include: (1) preoperative patient education, (2) nutrition screening, (3) smoking cessation when applicable, (4) use of topical anesthetic adjuncts, (5) facial nerve blocks, (6) postoperative analgesia, (7) preferential use of short-acting narcotics, (8) antibiotic administration, (9) use of elbow restraints, (10) early postoperative oral feeding and hydration, and (11) discharge planning.ResultsBetween April 2019 and March 2020, GSF operated on 126 patients with cleft lip from different age groups and 58.8% of them were less than 1 year of age. Three patients (2.4%) had delayed wound healing and one (0.8%) had postoperative bleeding. There were no cases of mortality, length of hospital stay did not exceed 1 postoperative day, and patients were able to tolerate fluids intake at discharge.ConclusionThe implementation of an ERAS protocol among patients undergoing cleft lip repair has shown to be highly effective in minimizing postoperative discomfort while reducing opioids use, decreasing the length of stay in hospital, and leading to early oral feeding resumption. The ERAS principles described carry increased relevance in the context of the ongoing COVID-19 pandemic and opioid crisis and can be safely applied in resource-constrained settings.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-15T05:35:14Z
      DOI: 10.1177/10556656221078744
       
  • Disruptive Therapy Using a Nonsurgical Orthodontic Airway Plate for the
           Management of Neonatal Robin Sequence: 1-Year Follow-up

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      Authors: HyeRan Choo, Rhona I. Galera, Karthik Balakrishnan, Hung-Fu C. Lin, HyoWon Ahn, Peter Lorenz, Rohit K. Khosla, Jochen Profit, Christian F. Poets, Janice S. Lee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      We recently published the 3-month follow-up of 2 neonates with Robin sequence whose mandibular hypoplasia and restricted airway were successfully treated with an orthodontic airway plate (OAP) without surgical intervention. Both infants were successfully weaned off the OAP after several months of continuous use. We present the course of OAP treatment in these patients with a focus on breathing, feeding, and facial growth during their first year of life. Both infants demonstrated stable mandibular projection, resolution of obstructive sleep apnea, and normal development.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-15T05:34:17Z
      DOI: 10.1177/10556656221076980
       
  • The Impact of Race and Ethnicity on Surgical Risk and Outcomes Following
           Palatoplasty: An Analysis of the NSQIP Pediatric Database

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      Authors: Jordan N. Halsey, Lindsey Asti, Richard E. Kirschner
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo determine how race and ethnicity affect palatoplasty 30-day outcomes.DesignRetrospective review.Patients/settingThe 2012 to 2019 National Surgical Quality Improvement Program (NSQIP) Pediatric database was used to identify patients ≤ 2 years who underwent primary palatoplasty. We compared demographics, comorbidities, and 30-day outcomes among different racial and ethnic groups. Logistic regression was used to determine independent risk factors for adverse events.Main outcome measuresIncreased risk for adverse events and postoperative surgical outcomes, including complications, readmission, and prolonged length of stay.ResultsA total of 8537 patients were identified in the database. African-American patients had the highest proportion of premature infants and infants with a BMI < 15% at the time of repair. Asian patients underwent palatoplasty at a later age compared to other races (12.7 months vs 11.7-12.1 months). Postoperatively, the odds of a complication were significantly higher in Asian patients (aOR = 1.73, 95% CI: 1.17-2.57) and other/unknown patients (aOR = 1.40, 95% CI: 1.05-1.86), but not among African American (aOR = 1.02, 95% CI: 0.70-1.47) or Hispanic (aOR = 0.93, 95% CI: 0.69–1.26) patients. Other/unknown patients were more likely to require postoperative ventilation (aOR = 2.34, 95% CI: 1.38-3.95). The odds of readmission were highest in Asian and other/unknown patients. African American, Hispanic, and other/unknown patients were more likely than Caucasian patients to be hospitalized> 2 days postoperatively.ConclusionThis study highlights ethnic differences in presentation and 30-day outcomes following palatoplasty. Further evaluation of disparities in cleft care should be performed to improve healthcare access and surgical outcomes.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-09T04:11:04Z
      DOI: 10.1177/10556656221078154
       
  • Socioeconomic Disparities in Cleft Lip Care

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      Authors: Christopher L. Kalmar, Elizabeth L. Malphrus, Mychajlo S. Kosyk, Zachary D. Zapatero, Jesse A. Taylor
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe purpose of this study was to utilize a multicenter dataset to elucidate whether socioeconomic factors were associated with access to cleft lip surgery, treatment by higher-volume providers, and family choice for higher-volume centers.DesignRetrospective cohort study.SettingHospitals participating in the Pediatric Health Information System.PatientsPrimary cleft lip repair performed in the United States between 2010 and 2020.OutcomesTravel distance, hospital volume, hospital choice.ResultsDuring the study interval, 8954 patients underwent unilateral (78.4%, n  =  7021) or bilateral (21.6%, n  =  1933) primary cleft lip repair. Patients with unilateral cleft lip were repaired significantly earlier if they were White (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-07T09:42:09Z
      DOI: 10.1177/10556656221078488
       
  • Bite Force Changes in Bilateral Cleft Lip and Palate Patients Before and
           After ALT-RAMEC Protocol

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      Authors: Sadia Shabbir, Anam Sattar, Qurat ul ain Sami, Babar Zia, Hana Pervez
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectivesTo evaluate the bite force (BF) changes in bilateral cleft lip and palate (BCLP) in contrast to the non BCLP children after ALT-RAMEC Facemask therapy.MethodsThe prospective study was carried out on a total of 60 subjects of age 6 to 13 years in a tertiary care hospital; treatment group, 30 children with BCLP and control group, 30 children without cleft lip and palate. Each cleft patient was received ALT-RAMEC Facemask treatment and their BF assessed at 4 different stages. Bite force taken before treatment, immediately, 3 months and 6 months after treatment. Bite force of the control group was taken at baseline to compare with the treatment group to demonstrate the difference in BF values. Independent t test and analysis of variance were used to perform statistical analysis.ResultsThere was a significant difference in BF between the treatment group (BCLP) 122.53 ± 8.64 N and the control group (non BCLP) 181.38 ± 18.84 N at baseline. After the Alt RAMEC protocol, changes in BF recorded over 3 and 6 months of therapy showed significant improvement. The mean values of BF at 3months and 6 months were 106.7 ± 9.3 N and 137.4 ± 9.5 N, respectively.ConclusionBite force was significantly improved after ALT-RAMEC protocol followed by facemask therapy in BCLP patients.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-07T05:53:06Z
      DOI: 10.1177/10556656211072730
       
  • Craniofacial Orthodontic Experience in CODA-Accredited Orthodontic
           Residency Programs

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      Authors: Manish Valiathan, Alexander DiMassa, Rebecca Petts, Rany Bous, Konstantinos Apostolopoulos, Tarek Elshebiny
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo evaluate orthodontic care for patients with craniofacial anomalies (CFA) by identifying orthodontic residents' preparedness to treat certain conditions and willingness to receive more training in CFA.Material and MethodsA 12-question survey was sent through the American Association of Orthodontics (AAO) organization to orthodontic residents. Questions were primarily designed to obtain information on the frequency with which they dealt with patients with CFA in their training, specific craniofacial conditions that orthodontic residents feel comfortable treating.ResultsA total of 150 participants out of 1066 responded. Of the 150 responses, 35% were first-year residents, 43% second year, and 22% were third-year residents. Thirty nine percent of residents saw 3 or more CFA patients during their residency followed by 24% that saw no patients with CFA. Forty five percent reported that 1 to 3 hours of lecture time was devoted to CFA per month. Sixty percent felt their training in CFA was not sufficient to feel comfortable treating these patients in practice. Specifically, 62% felt comfortable treating Down syndrome, 84% unilateral cleft lip and/or palate, and 64% bilateral cleft lip and/or palate, while the majority did not feel comfortable treating Pierre Robin sequence (68%), Cleidocranial dysplasia (65%), Crouzon syndrome (75%), Pfeiffer syndrome (80%), Treacher Collins syndrome (76%), Apert syndrome (76%), CHARGE syndrome (84%), and DiGeorge sequence (84%). Seventy eight percent of residents reported that they would like more training in treating craniofacial.ConclusionsOrthodontic residents did not feel comfortable treating patients with CFA. Majority of the residents felt that they would like to learn more about CFA.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-07T05:32:12Z
      DOI: 10.1177/10556656211059363
       
  • Epidemiology of Hospital Admissions for Craniosynostosis in Australia: A
           Population-Based Study

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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 trends, age, and sex-specific patterns of population hospital admissions with a diagnosis of craniosynostosis (CS) in Australia.Data SourcePopulation data for hospital separations (in-patient) from public and private hospitals (July 1996-June 2018) were obtained from the publicly available Australian Institute of Health and Welfare (AIHW) National Hospital Morbidity Database.Main outcome measuresThe outcome variables were hospital separation rates (HSR) (number of hospital separations divided by the estimated resident population [ERP] per year) and average length of stay (aLOS) (patient days divided by the number of hospital separations) with a diagnosis of CS. Trends in HSR and aLOS adjusted for age, sex, and type of CS were investigated by negative binomial regression presented as annual percent change (APC).ResultsIn 8057 admissions identified, we observed no significant change in the annual trend for HSR for the 22-year period. However, a marginal annual decrease of 1.6% (95% CI: -0.7, -2.4) in the aLOS was identified for the same time period. HSR were higher for males, infants, and single suture synostosis. aLOS was 3.8 days (95% CI: 3.8, 3.9) per visit, longer for syndromic conditions.ConclusionThere was a minor reduction in the average length of hospital stay for CS over the 22-year period potentially indicative of improved care. Population-level information on hospitalisations for rare craniofacial conditions can inform research, clinical, and surgical practice.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-02-07T05:31:53Z
      DOI: 10.1177/10556656221074215
       
  • Airway and Feeding Outcomes in Pierre Robin Sequence: A Comparison of
           Three Management Strategies

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      Authors: Ibrahim Khansa, Summer Aldabbeh, Gregory D. Pearson, Adriane Baylis, Lauren L. Madhoun, Anna Schoenbrunner, Mark Splaingard, Richard E. Kirschner
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundControversy remains regarding optimal management of Pierre Robin sequence (PRS). The goal of this study was to compare airway and feeding outcomes in infants with PRS who underwent surgical intervention, specifically mandibular distraction osteogenesis (MDO) or tongue-lip adhesion (TLA), or who had conservative management (CM) without surgery.MethodsAll consecutive patients treated for PRS at a pediatric academic medical center, with at least one year follow-up, were included. Patients who underwent tracheostomy as an index procedure were excluded. Patients were divided into those who underwent MDO, TLA or CM. Feeding status and data from initial and follow-up polysomnograms were collected. Comparisons between groups were made using the Kruskal-Wallis test, followed by Mann-Whitney pairwise comparison with a Bonferroni correction, when appropriate.Results67 neonates were included. 19 underwent TLA, 29 underwent MDO and 19 underwent CM. The proportions of syndromic patients were similar between groups. Patients undergoing CM had the lowest baseline AHI (9.1), but there were no significant differences between TLA (20.1) and MDO (25.4). At follow-up, the three groups had similar mean AHI (MDO 1.3, TLA 4.2, CM 4.5). A similar proportion of patients achieved AHI 5 or less (TLA 89.5%, MDO 96.6%, CM 84.2%). At one year, there were no significant differences in weight percentiles or in risk of failure-to-thrive between groups. One patient from the TLA group required a tracheostomy.ConclusionThe three treatment modalities achieved high airway and feeding success rates. All three modalities should have a place in the armamentarium of the craniofacial surgeon.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-31T10:57:50Z
      DOI: 10.1177/10556656221076345
       
  • Evaluation of Dimension and Bridging of Sella Turcica and Presence of
           Ponticulus Posticus in Individuals With and Without Cleft: A Comparative
           Study

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      Authors: Farzaneh Mosavat, Sarvin Sarmadi, Amirhesam Amini, Mohadeseh Asgari
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveSella turcica abnormalities such as sella turcica bridging (STB) have been observed frequently among patients with craniofacial anomalies. Ponticulus posticus (PP) is an important structure and its presence causes complications during surgeries. Due to the importance of these structures, this study aimed to evaluate the prevalence of STB, PP, and dimensions of the sella turcica in individuals with and without cleft.DesignA cross-sectional study.SettingOrthodontic and Radiology Departments in a public dental school, Tehran, Iran.PatientsEighty-three lateral cephalograms of individuals with cleft and ninety-two cephalograms of class I individuals without cleft.Main outcome measuresLength, depth, and maximum anterior–posterior diameter (APD) of sella turcica, presence of STB (classified as types 1, 2, and 3), and PP.ResultsIn the group with cleft, 19.3% of individuals had type I, 65.1%, type II, and 15.7%, type III STB. In the group without cleft, 15.2% had type I, 73.9%, type II, and 10.9%, type III STB. In the group with cleft, 65.1% of individuals had no PP, 24.1%, incomplete, and 10.8%, complete PP while in the group without cleft, 56.5% had no PP, 29 31.5%, incomplete, and 12%, complete PP.ConclusionThe sella turcica dimensions, STB, and PP prevalence were not significantly different between individuals with and without cleft. The sella turcica length and prevalence of STB and PP were not affected by age, sex, and cleft type. However, the depth and the APD were affected by age.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-31T10:57:41Z
      DOI: 10.1177/10556656221075935
       
  • Diagnostic Protocols for Alveolar Clefting and Barriers to Acquiring
           Imaging: A Survey of ACPA-Approved Cleft Teams in the United States

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      Authors: Kathryn Preston, Lucia Chen, Tyler Brennan, Barbara Sheller
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo report current diagnostic protocols, practices, and barriers related to imaging of alveolar clefting among American Cleft Palate-Craniofacial Association (ACPA)-approved cleft/craniofacial teams.MethodsAn electronic survey was sent to 162 ACPA-approved teams in the United States. Key items were team location, venue of orthodontic treatment, imaging modality(s) and access, barriers to imaging, billing, imaging protocols including team members involved in decisions pre- and post-alveolar bone grafting (ABG), and craniofacial fellowship status of team orthodontist(s).ResultsA total of 66 responses were received (40.7%). Responding teams were university-based (47%), hospital-based (42.4%), and independent clinics (10.6%). Orthodontic treatment for most patients was in private practice (53%). On-site 2-dimensional (2D) and 3-dimensional (3D) dental imaging capabilities were reported by 42% of teams; 29% have no on-site imaging. One or more barrier(s) to acquiring imaging were reported by 67%, with insurance challenges reported by 47%. Most teams bill medical payors for cleft-related dental imaging (58%). Pre- and post-ABG imaging was most frequently 3D (35% and 36%, respectively). Surgeons and orthodontists commonly evaluate ABG timing and outcome together (53%-65%). Periapical radiographs were included significantly more often in cleft imaging protocols by orthodontists with versus without fellowship training (P = .011, P = .04).ConclusionsBarriers to acquiring imaging are frequent. 3D is the most common imaging pre- and post-ABG. Our study endorses multi-level advocacy for improved medical insurance coverage of diagnostic cleft-related dental imaging to decrease barriers to providing timely care.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-31T03:10:03Z
      DOI: 10.1177/10556656221075938
       
  • Standard of Care for Patients With Cleft Lip and Palate in India—A
           Questionnaire-Based Study

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      Authors: Panchali Batra, Deborah Sybil, Arisha Izhar, Puneet Batra, Badri Thiruvenkatachari
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Background and ObjectivesA national survey of cleft teams was undertaken to evaluate the current standard of care for patients with cleft lip and palate (CLP) in India as a part of Cleft Care India study.DesignThis was a cross-sectional questionnaire-based study.MethodCleft teams across India attending the 19th Annual Conference of the Indian Society of Cleft Lip and Palate were invited to complete the questionnaire. The questionnaire consisted of 18 questions that included demographics, institutional details, patient protocols, surgical technique, rehabilitation facilities, and accessibility. The data are descriptively reported.ResultsA total of 112 centers completed the survey. Nongovernment organizations funded 87% of the cleft centers and 8% did not receive any funding. Only 39% of the centers had centralized cleft services providing multidisciplinary care. Speech therapy was provided either onsite or through referral at 90% of the centers, whereas audiology was provided only at 4% of centers. Feeding advice was routinely provided in 52% of centers. Millard technique was the most preferred technique for unilateral cleft lip repair (66%). The 2-flap technique (37%) and pharyngeal flap (48%) were the most common surgeries for cleft palate and pharyngoplasty, respectively. Although 54% of centers reported their patients to be interested in comprehensive care, 43% reported that their patients only wanted surgical correction.ConclusionsThere is wide diversity in access to cleft care and clinical practices across centers in India. Further work is needed to evaluate the quality of care by assessing outcomes of centers treating patients with CLP.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-31T03:10:02Z
      DOI: 10.1177/10556656221074212
       
  • A Clinical Report of the Complete Nasal Agenesis: Reconstruction of
           Congenital Arhinia and Review of the Literature

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      Authors: Etkin Boynuyogun, Gokhan Tuncbilek
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Congenital absence of the nose or arhinia is an exceptionally rare craniofacial malformation, and the pathophysiology of the arhinia is still unknown. Most arhinia patients can have difficulties with breathing and feeding due to the absence of the nose, nasal cavities, and associated problems. A 38-day-old female patient was referred to our clinic with arhinia. Physical examination revealed the complete agenesis of nasal structures as the nasal bones and vestibulum nasi. The region of the absent nose was flat and firm at palpation. Congenital arhinia may occur with other associated malformations such as ocular, ear, palate, and gonadal. Therefore, it is recommended to evaluate computed tomography/magnetic resonance imaging in the postnatal period. Additionally, a radiological evaluation will help nasal reconstruction by documenting changes in nasal and maxillary anatomy over time. Due to the limited number of arhinia cases presented, the surgical management of this condition has not been standardized. We presented the pyramid-shaped cartilage grafts for the nasal framework and an expanded paramedian forehead flap for the skin coverage for reconstruction of arhinia.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-24T12:22:01Z
      DOI: 10.1177/10556656221075939
       
  • Protraction Headgear Compliance and Orthognathic Surgery in Patients With
           Cleft Lip and Palate

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      Authors: Cleo Yi, Nilesh Shah, Bernard Costello, Jesse Goldstein, Anand Kumar, Joseph Losee, Lindsay Schuster
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveAssess cephalometric parameters and the need for orthognathic surgery (OS) and its relationship with compliance in protraction headgear (PHG).DesignRetrospective case series.SettingHospital cleft-craniofacial center.Patients23 patients with nonsyndromic cleft lip and palate and history of lip and palate repair.Interventions:Patients received PHG and orthodontic treatment. Compliant patients were compared to patients that were not. Protraction was applied with 170-gram elastics and patients were instructed to wear for at least 12 hours daily.Main outcome measuresCephalometric measurements at initial (T1), post-PHG (T2), and pre-surgical or post-orthodontic treatment (T3) of at least age 15 for females and 17 for males and the presence of OS were compared.Results83% (19) of patients reported compliance with therapy. Of those compliant, 68% (13) had OS and 32% (6) did not (P = .99). Inter-group comparisons at T1 between compliant and noncompliant showed no significant differences and the non-OS patients started with larger nasolabial angles (P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-24T12:21:42Z
      DOI: 10.1177/10556656221074890
       
  • A Rare Case of Glial Choristoma Arising from the Vomer in Association with
           Cleft Palate

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      Authors: Amrutha Varshini K, Anil Desai, BA Sahana, Niranjan Kumar
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Heterotopic neuroglial tissue represents normal glial tissue in an abnormal location distant from the central nervous system. It is a rare congenital condition and the majority of these lesions are diagnosed at birth or early childhood. We report a rare case scenario of a growth arising from the vomer associated with cleft palate. The origin of a glial choristoma from the midline of the nasal cavity in association with a cleft palate has not been reported in the literature. Complete surgical excision was performed prior to palatoplasty with no postoperative complications or evidence of recurrence.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-24T12:21:23Z
      DOI: 10.1177/10556656221074198
       
  • Surgical Repositioning of the Premaxilla: Incidence, Indications and
           Growth Study About a 189 Bilateral Cleft Lip  ±  Palate
           Population

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      Authors: Adrien Drilleaud, Pierre Corre, Jean-Philippe Perrin, Justine Loin, Thomas Durand, Jean-Michel Salagnac, Jean Delaire, Jacques-Marie Mercier, Helios Bertin
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveWe aimed to evaluate the rate of patient requiring Surgical Repositioning of the Premaxilla in a population carrying BCL  ±  P, retrieve age and operative indication. Our secondary objective was to present further facial growth characteristics.SettingsThis was a retrospective, single-center cohort study conducted in Nantes University Hospital, Oral and Maxillofacial Surgery department, tertiary cleft center.PatientsPatients with BCL  ±  P born between 1980 and 2019 treated at Nantes University Hospital were included.Main outcome measureOur primary outcome measure was the rate of patient requiring SRP.ResultsOver the whole period, 189 patients with BCL  ±  P were identified. Three patients (1,58%) underwent SRP. Patients who underwent SRP all had BCLP. SRP was performed during their primary dentition period. The indication for surgical repositioning was always premaxilla vertical overgrowth with an overbite over 10 mm. Facial growth features in the three patients were mostly comparable with a population carrying BCLP who had no premaxillary surgery.ConclusionOur results showed a low incidence of SRP. No SRP was necessary during early infancy (ie, before lip repair) or during adulthood. Surgical repositioning of the premaxilla is beneficial for patient with orthodontically uncorrectable vertical premaxillary excess, even more since facial development compared with other patients with BCLP appears comparable.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-24T12:21:14Z
      DOI: 10.1177/10556656211056807
       
  • Parental Satisfaction from Telemedicine in the Follow-up of Children
           Operated for Craniosynostosis during COVID-19 Pandemic

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      Authors: Evangelos G. Kilipiris, Frantisek Horn, Miroslava Kolnikova, Jose Vallejo Ochoa, Olga Matuskova, Drago Jelovac, Adam Stebel
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThe current study aimed to evaluate the satisfaction level of parents from telemedicine use in the long-term follow-up of children operated for craniosynostosis during the COVID-19 pandemic.DesignA cross-sectional 9-question survey analysis using Qualtrics survey software.SettingAn institutional study carried out at a national, tertiary level academic center in the Slovak Republic.PatientsAll patients operated for non-syndromic and syndromic craniosynostosis in our department, who participated in the virtual postoperative follow-up consultations from March 23, 2020, through July 2, 2021, were included in our series. They were enrolled 498 patients, with the parents of 256 children being responding to the survey.InterventionsThe survey remained open until July 16, 2021, two weeks after the last virtual consultations. It was delivered to the parents by e-mail to generate closed 5-point Likert scale responses.ResultsOverall, 72.3% of parents were satisfied with the telemedicine program, and 67.2% found it convenient. Collectively, 89.1% reported that the received instructions were helpful to them. However, only 18.7% of them answered that they would prefer telemedicine consultations in the future.ConclusionThe results of this study showed a high satisfaction from the parents during the virtual follow-up of children operated for craniosynostosis. However, despite their positive experience with telemedicine, the vast majority of parents stated that they would prefer in-person visits in the future.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-19T11:07:20Z
      DOI: 10.1177/10556656221074214
       
  • Labial Vestibular Flap Closure of the Cleft Palate Is Advantageous for
           Maxillary Development

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      Authors: Yukari Fujimoto, Susumu Tanaka, Koichi Otsuki, Tadashi Yamanishi, Emiko Isomura, Yusuke Yokota, Mikihiko Kogo
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveUsing labial vestibular flap was performed to close the primary alveolar and hard palate cleft at the second stage of early 2-stage closure surgery for unilateral cleft lip and palate for minimizing the damage to the maxillary periosteum. We analyzed maxillary development to clarify the influence of cleft palate surgery. Design: Retrospective longitudinal study in 5 years after primary palatal closure. Setting: Institutional study Patients: Study subjects included 214 patients with nonsyndromic complete unilateral cleft lip and palate who were consecutively treated in our clinic. Main Outcome: We used a 3D dental model scanner to assess maxillary development in patients aged 3 months to 5 years after using either the conventional pushback method (PB) (51 cases) or 2-stage closure (Local palatal flap closure: LF [67 cases] and Labial vestibular flap closure: VF [96 cases]). Results: Comparing the measurement results, the major axis of maxilla, width, intercanine distance, and intermolar distance was significantly larger in the LF group compared to the PB group. After the age of 3, the cleft side of VF group had grown significantly to compare with LF group in width. It was also confirmed that the inserted labial mucosal flap itself grew. Enlargement of the labial mucosal flap was observed at all sites except the canine. Conclusion: Good maxillary growth occurred in the following order: VF groups> LF group> PB group. Poor growth was correlated with the extent of periosteal damage during surgery and the degree of postoperative bone surface exposure.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-19T11:07:05Z
      DOI: 10.1177/10556656211065944
       
  • Presurgical Orthopedic Treatment of a 3-Year-Old Child with Unoperated
           Complete Bilateral Cleft Lip and Palate and Protruding Premaxilla

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      Authors: Julija Radojicic, Branislav Trifunovic, Tatjana Cutovic, Aleksandra Radojicic
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      Unoperated patients with bilateral complete cleft lip and palate (BCCLP), who do not receive orthopedic treatment in infancy and present with significant sagittal excess of the premaxilla, are among the most difficult patients to treat. Delay in primary reconstruction reduces the elastic characteristics of the tissues. The resulting rigidity of bony structures accompanied by the wide alveolar and palatal clefts associated with sagittal excess of the premaxilla pose a great challenge for both surgeon and orthodontist. There is no unique protocol for treatment of these conditions. This paper describes the use of orthopedic treatment in an unoperated 3-year-old male child with BCCLP and a protrusive premaxilla whose appearance and function were both severely affected. A modified Meazzini technique, as described in operated BCCLP patients with a downward-displaced premaxilla, was used. The device itself consists of the 2 independent components mutually connected by a 17 × 25 steel arch. The first is an active fan-type expander. The second consists of an acrylic cap covering the premaxilla and 3 brackets used for the steel arch application. They are connected by a rectangular steel 17 × 25 archwire. Reduction of the premaxillary sagittal excess of is carried out with an elastic chain. The use of this orthopedic appliance is a reliable and valuable presurgical treatment in the therapy of children with BCCLP and protrusive premaxilla whose orthopedic and surgical treatment have been delayed.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-19T03:03:33Z
      DOI: 10.1177/10556656221074190
       
  • Disparities in Timing of Alveolar Bone Grafting and Dental Reconstruction
           in Patients With Clefts

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      Authors: Ryan K. Badiee, Stephen C. Yang, Andre Alcon, Andrew C. Weeks, Glenn Rosenbluth, Jason H. Pomerantz
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveThis study sought to identify disparities in the timing of alveolar bone grafting (ABG) surgery and the replacement strategy for missing maxillary lateral incisors for patients with clefts.DesignA retrospective record review identified patients who underwent ABG. Multivariable regression analyzed the independent contribution of each variable.SettingThis institutional study was performed at the University of California, San Francisco.ParticipantsPatients who presented under age 12 and underwent secondary ABG between 2012 and 2020 (n = 160).Main Outcome MeasuresThe age at secondary ABG and the recommended dental replacement treatment for each patient, either dental implantation or canine substitution.ResultsThe average age at ABG was 10.8 ± 2.1 years, 106 (66.3%) patients were not White, and 80 (50.0%) had private insurance. Independent predictors of older age at ABG included an income below $ 50 000 as estimated from ZIP code (β = 15.0 months, 95% CI, 5.7-24.3, P = .002) and identifying as a race other than White (β = 10.1 months, 95% CI, 2.1-18.0, P = .01). After ABG, patients were more likely to undergo dental implantation over canine substitution if they were female (odds ratio [OR] = 4.3, 95% CI, 1.3-17.1, P = .02) or had private insurance (OR = 12.5, 95% CI, 2.2-143.2, P = .01).ConclusionsPatients who were low-income or not White experienced delays in ABG, whereas dental implantation was more likely to be recommended for patients with private insurance. Understanding the sources of disparities in dental reconstruction of cleft deformities may reveal opportunities to improve equity.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-19T03:03:14Z
      DOI: 10.1177/10556656211073049
       
  • Morphology of the Occipital Bones and Foramen Magnum Resulting From
           Premature Minor Suture Fusion in Crouzon Syndrome

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      Authors: Ryan G. Layton, Joshua F. Pontier, Griffin P. Bins, Brandon J. Sucher, Christopher M. Runyan
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo identify skull-base growth patterns in Crouzon syndrome, we hypothesized premature minor suture fusion restricts occipital bone development, secondarily limiting foramen magnum expansion.DesignSkull-base suture closure degree and cephalometric measurements were retrospectively studied using preoperative computed tomography (CT) scans and multiple linear regression analysis.SettingEvaluation of multi-institutional CT images and 3D reconstructions from Wake Forest’s Craniofacial Imaging Database (WFCID).PatientsSixty preoperative patients with Crouzon syndrome under 12 years-old were selected from WFCID. The control group included 60 age- and sex-matched patients without craniosynostosis or prior craniofacial surgery.InterventionsNoneMain Outcomes Measure(s)2D and 3D cephalometric measurements.Results3D volumetric evaluation of the basioccipital, exo-occipital, and supraoccipital bones revealed decreased growth in Crouzon syndrome, attributed solely to premature minor suture fusion. Spheno-occipital (β = −398.75; P 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-19T03:02:54Z
      DOI: 10.1177/10556656211072762
       
  • Tympanostomy Tubes: Are They Necessary' A Systematic Review on
           Implementation in Cleft Care

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      Authors: Eloise Stanton, Katelyn Kondra, Indraneel Brahme, Sasha Lasky, Naikhoba C.O. Munabi, Christian Jimenez, Laya Jacob, Mark M. Urata, Jeffrey A. Hammoudeh, William P. Magee
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo understand the indication for and the effects of early ventilation tube insertion (VTI) on hearing and speech for patients with cleft lip and/or palate (CLP).DesignWe conducted a Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA)-guided systematic review of relevant literature.SettingSetting varied by geographical location and level of clinical care across studies.Patients, ParticipantsPatients with CLP who underwent VTI were included.InterventionsNo interventions were performed.Main Outcome Measure(s)Primary outcome measures were hearing and speech following VTI. Secondary outcome measures were tube-related and middle ear complications. Early VTI occurred before or at time of palatoplasty while late VTI occurred after palatoplasty.ResultsTwenty-three articles met inclusion criteria. Articles varied among study design, outcome measures, sample size, follow-up, and quality. Few studies demonstrated support for early VTI. Many studies reported no difference in hearing or speech between early and late VTI. Others reported worse outcomes, greater likelihood of complications, or needing repeat VTI following early tympanostomy placement. Several studies had significant limitations, including confounding variables, small sample size, or not reporting on our primary outcome.ConclusionsNo consistency was found regarding which patients would benefit most from early VTI. Given the aforementioned variability and sub-optimal methodologies, additional studies are warranted to provide stronger evidence regarding VTI timing in cleft care.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-19T03:02:47Z
      DOI: 10.1177/10556656211067901
       
  • Maternal Stressful Life Events During the Periconceptional Period and
           Orofacial Clefts: A Systematic Review and Meta-Analysis

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      Authors: Christina Tran, Andrew A. Crawford, Alexander Hamilton, Clare E. French, Yvonne Wren, Jonathan Sandy, Gemma Sharp
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveTo assess whether women who experience stressful life events during the periconceptional period are at higher risk of giving birth to a baby with an orofacial cleft (OFC).DesignSystematic review and meta-analysis of studies reporting the proportion of babies born with OFC to mothers exposed and unexposed to population-level or personal-level stressful life events during the periconceptional period. Six electronic databases were searched from inception to August 2020. Risk of bias was assessed using the Newcastle-Ottawa scale. Odds ratios (ORs) for the odds of OFC in babies of exposed mothers relative to unexposed controls were extracted and/or calculated. Random effects meta-analysis was undertaken, stratified by cleft subtype.ResultsOf 12 eligible studies, 8 examined experience of personal events and 4 examined population-level events. Studies demonstrated low-moderate risk of bias and there was indication of publication bias. There was some evidence that personal stressful life events were associated with greater odds of cleft lip and/or palate (six studies, OR 1.63, 95% confidence interval (CI) 1.16, 2.30, P = 0.001) and cleft palate only (six studies, OR 1.45, 95% CI 1.02, 2.06, P = 0.04). Population-level events were associated with higher odds of OFC in studies that did not specify subtype (three studies, OR 1.64, 95% CI 1.19, 2.25, P = 0.002), but subtype stratified analyses were underpowered. Heterogeneity was high.ConclusionsLimited evidence indicated a weak positive association between maternal stressful life events during the periconceptional period and risk of OFC in the offspring, but further studies with greater consistency in research design are needed.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-11T03:13:09Z
      DOI: 10.1177/10556656211045553
       
  • Facial Nerve Dysfunction After Mandibular Distraction Osteogenesis in
           Patients with Robin Sequence: A Systematic Review and Meta-Analysis

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      Authors: Elina Kapoor, Esperanza Mantilla-Rivas, Md Sohel Rana, Marudeen Aivaz, Daniela Duarte-Bateman, Joseph M. Escandón, Hannah R. Crowder, Monica Manrique, Gary F. Rogers, Albert K. Oh
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveRobin Sequence (RS), characterized by micrognathia, glossoptosis, and upper airway obstruction, is an increasingly recognized diagnosis. An effective surgical intervention is mandibular distraction osteogenesis (MDO). This study analyzes published evidence regarding facial nerve dysfunction (FND) associated with MDO.Design and Setting According to PRISMA guidelines, a systematic review was carried out with databases queried in June 2019 using MESH terms, or equivalent terms, as follows: “distraction osteogenesis” and “Robin Sequence”. A review of original Spanish and English articles, were included. Outcome measures included the prevalence of FND; the affected branches; the rate of permanent vs. transient FND; the use of an internal vs. external device; the daily distraction rate; and finally, the overall distraction length. Subsequently, a meta-analysis was conducted to collate results regarding the prevalence of FND and the factors associated with it. ResultsOf 239 unique studies identified, 19 studies with 729 patients met inclusion criteria; 52 patients developed FND after MDO. A random-effects meta-analysis yielded a pooled prevalence of FND of 6.40%, with moderately heterogeneous studies (I2 = 41%, τ2 = 0.006). Marginal mandibular nerve involvement was most commonly noted. Nine studies reported transient FND, six permanent, one both, and two unspecified. Internal distractors were used in 8 studies and external in 3 and both in 2. Distraction rate was 1.00 to 2.00 mm/day and total distraction length ranged from 13.00 to 22.3 mm. Sample size was the only parameter inversely associated with rate of FND (p = 0.04).ConclusionThis analysis of FND associated with MDO for patients with RS demonstrates a lack of consistent documentation. MDO-associated FND does not appear to be uncommon, and permanent dysfunction can occur. This review underscores the importance of thorough documentation to elucidate the mechanism of FND.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-10T12:47:47Z
      DOI: 10.1177/10556656211070728
       
  • Adapting Elements of Cleft Care Protocols in Low- and Middle-income
           Countries During and After COVID-19: A Process-driven Review With
           Recommendations

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      Authors: Matthew Fell, Michael Goldwasser, B.S. Jayanth, Rui Manuel Rodrigues Pereira, Christian Tshisuz Nawej, Rachel Winer, Neeti Daftari, Hugh Brewster, Karen Goldschmied, Collaborators: Fernando Almas, Mekonen Eshete, George W. Galiwango, Larry H. Hollier, Akhter Hussain, Lun-Jou Lo, Paul Salins, Debbie Sell, Amanuel Tafase, Ronald M. Zuker
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveA consortium of global cleft professionals, predominantly from low- and middle-income countries, identified adaptations to cleft care protocols during and after COVID-19 as a priority learning area of need.DesignA multidisciplinary international working group met on a videoconferencing platform in a multi-staged process to make consensus recommendations for adaptations to cleft protocols within resource-constrained settings. Feedback was sought from a roundtable discussion forum and global organizations involved in comprehensive cleft care.ResultsFoundational principles were agreed to enable recommendations to be globally relevant and two areas of focus within the specified topic were identified. First the safety aspects of cleft surgery protocols were scrutinized and COVID-19 adaptations, specifically in the pre- and perioperative periods, were highlighted. Second, surgical procedures and cleft care services were prioritized according to their relationship to functional outcomes and time-sensitivity. The surgical procedures assigned the highest priority were emergent interventions for breathing and nutritional requirements and primary palatoplasty. The cleft care services assigned the highest priority were new-born assessments, pediatric support for children with syndromes, management of acute dental or auditory infections and speech pathology intervention.ConclusionsA collaborative, interdisciplinary and international working group delivered consensus recommendations to assist with the provision of cleft care in low- and middle-income countries. At a time of global cleft care delays due to COVID-19, a united approach amongst global cleft care providers will be advantageous to advocate for children born with cleft lip and palate in resource-constrained settings.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-04T03:47:04Z
      DOI: 10.1177/10556656211069827
       
  • The Cleft Lip Education with Augmented Reality (CLEAR) VR Phase 2 Trial: A
           Pilot Randomized Crossover Trial of a Novel Patient Information Leaflet

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      Authors: Steven John Lo, Paul Chapman, David Young, David Drake, Mark Devlin, Craig Russell
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      BackgroundThe Cleft Lip Education with Augmented Reality (CLEAR) project centers around the use of augmented reality (AR) in patient leaflets, as a visual means to overcome the “health literacy” gap. This trial followed Virtual Reality (VR CORE) guidelines for VR Phase 2 (Pilot) trials.MethodsParticipants included families of children treated for Cleft Lip and Palate at the Royal Hospital for Children, Glasgow. Interventions were AR leaflet or Traditional Leaflet. Objectives were to calculate sample sizes, assess outcome instruments, trial design, and acceptability to patients. Primary outcome measure was Mental Effort Rating Scale, and secondary outcomes were Patient Satisfaction (Visual Analogue Scale), Usefulness Scale for Patient Information Material (USE) scale, and Instructional Materials Motivation Survey (IMMS). Randomization was by block randomization. The trial was single blinded with assessors blinded to group assignment.Results12 Participants were randomized, with crossover design permitting analysis of 12 per group. Primary outcome with Mental Effort Rating Scale indicated higher mental effort with Traditional compared to AR Leaflet (4.75 vs 2.00, P = .0003). Secondary outcomes for Satisfaction were Traditional 54.50 versus AR 93.50 (P = .0001); USE scale 49.42 versus 74.08 (P = .0011); and IMMS 112.50 versus 161.75 (P = .0003). Subjective interviews noted overwhelmingly positive patient comments regarding the AR leaflet. Outcome instruments and trial design were acceptable to participants. No harms were recorded.ConclusionsThe CLEAR pilot trial provides early evidence of clinical efficacy of AR leaflets in patient education. It is hoped that this will provide a future paradigm shift in the way patient education is delivered.
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-04T03:46:50Z
      DOI: 10.1177/10556656211059709
       
  • Risk of Malocclusion Among Patients Undergoing Single-Stage Versus
           Two-Stage Cleft Palate Repair

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      Authors: Hilary McCrary, Vanessa Torrecillas, Sarah Hatch Pollard, Dave S. Collingridge, Duane Yamashiro, Jonathan R. Skirko
      Abstract: The Cleft Palate-Craniofacial Journal, Ahead of Print.
      ObjectiveEvaluate impact of single-stage versus staged palate repair on the risk of developing malocclusion among patients with cleft palate (CP).DesignRetrospective cohort study 2000–2016SettingAcademic, tertiary children’s hospital.PatientsPatients undergoing CP repair between 1999–2015.InterventionsCP repair, categorized as either single-stage or staged.Main Outcome MeasureTime to development of Class III malocclusion.Results967 patients were included; 60.1% had a two-stage CP repair, and 39.9% had single-stage. Malocclusion was diagnosed in 28.2% of patients. In the model examining all patients at ≤5 years (n = 659), patients who were not white had a higher risk of malocclusion (HR 2.46, p = 0.004) and staged repair was not protective against malocclusion (HR 0.98, p = 0.91). In all patients>5 years (n = 411), higher Veau classification and more recent year of birth were significantly associated with higher hazard rates (p 
      Citation: The Cleft Palate-Craniofacial Journal
      PubDate: 2022-01-04T03:46:27Z
      DOI: 10.1177/10556656211044944
       
 
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