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Abstract: Introduction Vagus nerve stimulation (VNS) is an adjunctive treatment in children with intractable epilepsy. When lead replacement becomes necessary, the old leads are often truncated and retained and new leads are implanted at a newly exposed segment of the nerve. Direct lead removal and replacement are infrequently described, with outcomes poorly characterized. We aimed to describe our experience with feasibility of VNS lead removal and replacement in pediatric patients. Methods Retrospective review examined 14 patients, at a single, tertiary-care, children’s hospital, who underwent surgery to replace VNS leads, with complete removal of the existing lead from the vagus nerve and placement of a new lead on the same segment of the vagus nerve, via blunt and sharp dissection without use of electrocautery. Preoperative characteristics, stimulation parameters, and outcomes were collected. Results Mean age at initial VNS placement was 7.6 years (SD 3.5, range 4.5–13.4). Most common etiologies of epilepsy were genetic (5, 36%) and cryptogenic (4, 29%). Lead replacement was performed at a mean of 6.0 years (SD 3.8, range 2.1–11.7) following initial VNS placement. Reasons for revision included VNS lead breakage or malfunction. There were no perioperative complications, including surgical site infection, voice changes, dysphagia, or new deficits postoperatively. Stimulation parameters after replacement surgery at last follow-up were similar compared to preoperatively, with final stimulation parameters ranging from 0.25 mA higher to 1.5 mA lower to maintain baseline seizure control. The mean length of follow-up was 7.9 years (SD 3.5, range 3.1–13.7). Conclusion Removal and replacement of VNS leads are feasible and can be safely performed in children. Further characterization of surgical technique, associated risk, impact on stimulation parameters, and long-term outcomes are needed to inform best practices in VNS revision. PubDate: 2022-11-25
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Abstract: Background Longstanding overt ventriculomegaly in adults (LOVA) is a new form of progressive hydrocephalus characterized by onset in early childhood and gradual progression into adulthood. Patients with LOVA are usually asymptomatic in childhood. The diagnosis of LOVA in adolescence has not been reported. Case report A patient with macrocephaly and mild ventriculomegaly from infancy developed headache exacerbation and cognitive dysfunction at the age of 11 years. Brain magnetic resonance imaging showed mild tri-ventriculomegaly with no radiological aggravation compared to imaging at the age of 8 years. No papilledema was observed. Drainage of 15 ml of spinal fluid via a lumbar puncture relieved the headache and cognitive dysfunction. Based on repeated improvements in cognitive function and headaches after spinal fluid drainage, we diagnosed the patient with LOVA with symptom onset in early adolescence. A ventriculoperitoneal shunt was placed, and the headaches disappeared completely. The full-scale intellectual quotient, verbal comprehension, and working memory improved significantly. Conclusions LOVA may manifest as early as adolescence. The clinical presentation, age, clinical, radiological features, and management vary, and a spinal tap exam is useful for diagnosing LOVA, even in children. The spinal tap exam may be indicated in children with longstanding ventriculomegaly and deteriorating neurological symptoms to diagnose this “treatable intellectual disability.” PubDate: 2022-11-24
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Abstract: Purpose Sacrococcygeal teratoma (SCT) is a rare congenital tumor originating from a variant of extragonadal germ cell neoplasm. Herein, we present three cases of neonatal SCTs undergoing surgical resection and reconstruction. We also review the literature to discuss the importance of proper perinatal management and timely surgical intervention depending on the tumor type and maturity to prevent malignant transformation and recurrence and ensure functional outcomes. Methods and results Three cases of SCT were retrospectively analyzed. All infants underwent complete surgical resection of the SCT and coccygectomy during their neonatal/infancy period, followed by pelvic floor and buttock reconstruction while minimizing buttock contour deformity and undesirable skin scar. Two of the cases were histopathologically diagnosed as predominantly cystic mature teratomas of Altman types I and IV, and the third was a mature teratoma of Altman type II. There were no complications or tumor recurrence during the average follow-up period of 5.3 years. Conclusions This case-based review highlights the role of multidisciplinary team approaches, including prenatal monitoring, oncologic resection, and adequate reconstruction according to the type of tumor and anomaly. Optimal perinatal evaluation allows promising oncologic and functional outcomes in terms of timely intervention to eliminate tumor recurrence and malignant transformation. Complete oncologic surgical resection of SCTs should also include proper functional preservation strategies, such as the reconstruction of the pelvic floor, cosmetic buttock contouring, and preservation of bladder and bowel sphincter function. PubDate: 2022-11-24
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Abstract: Abstract Dr. Boop is the president of the 48th annual meeting of the ISPN. He has served Semmes-Murphey Clinic as a member of the Board of Directors, Chief of the pediatric neurosurgery division, and as pediatric neurosurgery fellowship director. He has held the JT Robertson endowed chair from The University of Tennessee Health Science Center (UTHSC) Department of Neurosurgery residency training program and the St. Jude endowed chair in pediatric neurosurgery where he is currently a member of the St. Jude Global Program. He is currently Emeritus Chair at UTHSC. PubDate: 2022-11-23
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Abstract: Primary Ewing’s sarcoma originating from the calvaria bone and/or underlying Dural involvement has been reported relatively rarely in the literature. Those originating from the dura and invading the bone above it in both directions, both towards the brain parenchyma and via the dura, are even rarer. Case description We present a case of a 14-year-old girl with no known focal neurological deficit who presented with the complaint of vertigo for only 2 months. In neuroradiological examination, the left frontoparietal region of the brain showed the presence of a tumor originating from the dura, invading both bone and brain parenchyma. No other tumor location was discovered after radiological examination. Since the patient had a shift in the brain and progressive loss of strength on the right side, the patient was taken to surgery for tumor excision. The frozen result sent per-operatively was consistent with a round blue cell tumor. Adjuvant chemotherapy treatment was given to the patient after the definitive pathology report was compatible with Ewing’s sarcoma. Conclusion The patient had an uneventful neurological recovery without permanent neurological deficit. When the patient was kept under close clinical and radiological surveillance 1 year after the operation, no recurrence of the disease was observed. Bone marrow biopsy results and pet computerized tomography results confirmed the case of primary intracerebral Ewing sarcoma. This case illustrates an extremely rare location of primary Ewing’s sarcoma with a set of clinical signs and symptoms extremely rare for this location of this rare disease entity. PubDate: 2022-11-23
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Abstract: Abstract Chordomas are rare malignant bone tumours that develop from the ectopic remnants of the embryonic notochord. In contrast to adults, the majority in children under 16 present intra-cranially (63%). In 2006, we reported the youngest case of a large clival chordoma, a 15-week old baby, the second case to present without skull base involvement and the fourth case of chordoma in a patient with tuberous sclerosis (TS) Kombogiorgas (Childs Nerv Syst 22(10):1369–1374, 2006). In this report, we provide an update on this patient’s journey through a range of therapeutic options and summarize an update of the literature, since 2006, for this patient group. PubDate: 2022-11-22
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Abstract: Background Aggressive vertebral hemangiomas are rare tumors in children, usually occurring in the thoracic spine that can cause significant neurological morbidity. They are technically difficult to treat with significant risk of blood loss during surgery. Methods We describe a case of aggressive vertebral hemangioma managed in our institution. We performed a literature review of reported cases of aggressive vertebral hemangiomas in pediatric age group. We discuss the clinical presentation, diagnosis, and management of these lesions. Results We identified 23 cases of aggressive vertebral reported in children. Neurodeficit was the most common presentation, and the most common location was the thoracic spine. Surgery was the most common modality of treatment. All the patients reported in literature had improvement in their symptoms after treatment. Conclusion Although technically challenging, aggressive vertebral hemangiomas have a good outcome after treatment. Treatment should be tailored to the individual patient. Further studies are needed to determine the optimum treatment strategy. PubDate: 2022-11-22
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Abstract: Purpose Cavernous malformations (CM) are central nervous system lesions characterized by interlaced vascular sinusoids coated with endothelial cells without intervening parenchyma. Magnetic resonance imaging-guided laser interstitial thermal therapy (MRIgLITT) is a minimally invasive treatment modality that can precisely treat pathologic cerebral tissue, making it an effective alternative for the management of cavernomas. We describe the outcomes of a series of pediatric patients with cavernous brain malformations treated with MRIgLITT between 2014 and 2018 at our institution. Methods We retrospectively analyzed 11 cavernomas in 6 pediatric patients treated with MRIgLITT. Both the Visualase System® and/or Neuroblate® systems were used. A variation of the surgical technique on the application of the laser was developed. Post-ablation MRIs were obtained to assess ablated areas. Results A total of 11 cavernomas in 6 patients were treated with MRIgLITT. Median age was 15 years (12 to 17 years); 75% were males. Presenting symptoms were headache (75%) and seizures (25%). Two patients presented with multiple CMs. All lesions in this study were supratentorial (cerebral hemispheres 81.8%, corpus callosum 9.1%, basal ganglia 9.1%). Our surgical technique was well-tolerated, with no significant adverse events observed. Hospital stay for all patients was less than 48 hours. Conclusion MRIgLITT is an effective minimally invasive technique for the treatment of pediatric CMs. It represents a useful and safe tool, when other therapeutic alternatives may represent a greater risk of surgical morbidity. PubDate: 2022-11-22
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Abstract: Introduction While operative intervention for Chiari malformation type I (CMI) with syringomyelia is well established, there is limited data on outcomes of intraoperative neuromonitoring (IONM). This study sought to explore differences in procedural characteristics and their effects on postoperative readmission rates. Methods The Nationwide Readmission Database was queried from 2010 to 2014 for patients ≤ 18 years of age with CMI and syringomyelia who underwent cranial decompression or spinal decompression. Demographics, hospital characteristics, and outcomes were analyzed. Results Over the 5-year period, 2789 patients were identified that underwent operative treatment for CMI with syringomyelia. Mean age was 10 ± 4 years with 55% female. During their index hospitalization 14% of the patients had IONM. Patients receiving IONM had no significant difference in Charleston Comorbidity Index ≥ 1 (16% vs. 15% without, p = 0.774). IONM was more often used in those with private insurance (63% vs. 58% without, p = 0.0004) and less likely in those with Medicaid (29% vs. 37% without, p = 0.004). Patients receiving IONM were more likely to have a postoperative complication (23% vs 17%, p = 0.004) and were more likely to have hospital lengths of stay > 7 days (9% vs. 5% without, p = 0.005). Readmission rates for CMI were 9% within 30 days and 15% within the year. The majority (89%) of readmissions were unplanned. 25% of readmissions were for infection and 27% of readmissions underwent a CMI reoperation. The 30-day readmission rate was higher for those with IONM (12% vs. 8% without, p = 0.010). Median cost for hospitalization was significantly higher for patients with IONM ($26,663 ($16,933–34,397)) vs. those without ($14,577 ($11,538–18,392)), p < 0.001. Conclusion The use of intraoperative neuromonitoring for operative repair of CMI is associated with higher postoperative complications and readmissions. In addition, there are disparities in its use and increased cost to the healthcare system. Further studies are needed to elucidate the factors underlying this association. PubDate: 2022-11-21
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Abstract: Purpose The objective of this literature review was to provide a comprehensive and up-to-date overview of the current understanding of the genetic etiology for non-syndromic sagittal craniosynostosis. Methods Using the PubMed database and Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA), we systematically reviewed relevant records on germline genetics in children with non-syndromic sagittal craniosynostosis. Results Two hundred two records were identified, of which 25 were included following title and abstract screening and subsequent full-text review. The 25 records in combination included 829 children with non-syndromic sagittal craniosynostosis. A likely pathogenic or pathogenic germline variant was reported for 9.8% of the 827 patients for whom germline genetic testing was performed. The reported variants were distributed across 50 different genes, with more than one variant detected in 13 genes. Conclusion Based on the existing literature, genetic predisposition is likely to play a role in at least 9% of children with non-syndromic sagittal craniosynostosis. Future studies will benefit from international consensus in terms of diagnostic nomenclature and a higher level of standardization across study methodologies and bioinformatic approaches. PubDate: 2022-11-19
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Abstract: Abstract The abusive head trauma (AHT) is a form of child abuse and is a frequent entity all over de world. It is particularly unique among medical diagnoses because of the legal implications imposed by the diagnosis. Therefore, it has been the subject of much legal controversy over the decades. Knowledge of the clinical signs and imaging findings of abusive head trauma is vitally important for early diagnosis. An oriented anamnesis, as well as a complete physical examination and obtaining adequate images of the central nervous system, play a significant role in confirming the presumptive diagnosis. The interdisciplinary approach (pediatricians, neurosurgeons, neuroradiologists, social workers, and other specialists) is the key in the management of these patients. The purpose of this article is to familiarize the pediatric neurosurgeon with some of the more common medicolegal issues surrounding AHT as well as to discuss legal commitments and ethical obligations of the neurosurgeon in Argentina (South America) based on 2 clinical cases. PubDate: 2022-11-18
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Abstract: Purpose Patients with mucopolysaccharidosis type IVA (MPS IVA) have many risk factors for myelopathy and paresis. These are spinal cord compression, bone stenosis, and soft tissue thickening with ligament laxity, deformity, odontoid hypoplasia, and atlantoaxial instability. Although most patients with MPS IVA appear generally healthy at birth, patients often show skeletal deformities within a few years. Surgical indications are difficult to determine. Historically, many physicians have used prophylactic decompression and fusion in young, asymptomatic MPS IVA patients to prevent cord compression. Although spinal cord decompression is usually required at the craniocervical junction in patients with MPS IVA, decompression may be required at other spinal cord levels as well. There is a risk of developing neurological damage during surgery. The most common causes are ischemia secondary to cardiac output deteriorated in the prone position or due to artery damage, and local trauma due to neck movements or traction while bringing the patient to the prone position. Neurophysiological monitoring is very important during surgery to reduce the risk of neurological damage in spinal cord surgery. In this case report, a case with loss of lower extremity neuromonitorization motor evoked potential (MEP) responses in the early period of surgery without any intervention to the craniocervical junction after prone positioning will be presented. PubDate: 2022-11-18
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Abstract: Introduction Choroidal fissure cysts (CFC) are usually an incidental finding on imaging and lead to morbidity only in rare cases. The aim of this study was to present the case of a patient with a giant CFC and its treatment. Case report The patient was a male infant of 9 days of life that presented with symptoms of intracranial hypertension. A CFC measuring 56 × 70 × 86 mm was diagnosed. Endoscopic fenestration of the cyst was performed; however, the communication closed thereafter. A reservoir catheter was inserted, which subsequently became infected and was then replaced by a ventriculoperitoneal shunt. Discussion The choroidal fissure is a C-shaped cleft located between the fornix and the thalamus separated from the temporal horn by the layer of tela choroidea. The pathophysiology of CFC is still unclear. In our case, initial endoscopic treatment was decided on to avoid shunt-related complications. Currently, there are no guidelines for the best surgical treatment for CFC. Conclusion We describe an atypical case of CFC and analyze the surgical treatment performed. Although endoscopic treatment is recommended for CFC, currently no guidelines exist regarding the best management of the entity. PubDate: 2022-11-17
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Abstract: Background Variables interacting to predict outcomes following spring-mediated cranioplasty (SMC) for non-syndromic craniosynostosis, including spring parameters and calvarial thickness, are poorly understood. This study assessed interactions between spring parameters and calvarial thickness to predict changes in cephalic index (CI) following SMC. Methods Patients undergoing SMC for non-syndromic sagittal craniosynostosis at our institution between 2014 and 2021 were included. Calvarial thickness was determined from patient preoperative CTs using Materalise Mimics at 27 points in relation to the sagittal suture. Linear mixed effects models were used to determine interactions between anterior, middle, and posterior calvarial thickness with spring force and length. Results Sixty-nine patients undergoing surgery at mean age 3.7 months were included in this study. Stronger posterior spring force interacted with thinner posterior calvarial thickness to predict greater changes in CI at 3 months postoperatively (p = 0.022). When evaluating spring force and calvarial thickness set distances from the sagittal suture, stronger posterior spring force interacted with thinner posterior calvarial thickness 5 mm (p = 0.043) and 10 mm (p = 0.036) from the sagittal suture to predict changes in CI. Interactions between spring parameters and calvarial thickness in the anterior and middle positions did not significantly predict changes in CI. Conclusions Stronger posterior spring force interacted with thinner posterior calvaria to predict greater changes in CI 3 months following SMC for non-syndromic sagittal craniosynostosis. These results suggest dynamic interactions between several variables may impact outcomes following SMC. PubDate: 2022-11-17
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Abstract: Purpose The purpose of the study was to better understand the clinical course and impact of tethered cord release surgery on patients who have previously undergone open spinal dysraphism closure in utero. Methods This is a single-center retrospective observational study on patients undergoing tethered cord release after having previously had open fetal myelomeningocele (MMC) closure. All patients underwent tethered cord release surgery with a single neurosurgeon. A detailed analysis of the patients’ preoperative presentation, intraoperative neuromonitoring (IONM) data, and postoperative course was performed. Results From 2009 to 2021, 51 patients who had previously undergone fetal MMC closure had tethered cord release surgery performed. On both preoperative and postoperative manual motor testing, patients were found to have on average 2 levels better than would be expected from the determined anatomic level from fetal imaging. The electrophysiologic functional level was found on average to be 2.5 levels better than the anatomical fetal level. Postoperative motor levels when tested on average at 4 months were largely unchanged when compared to preoperative levels. Unlike the motor signals, 46 (90%) of patients had unreliable or undetectable lower extremity somatosensory evoked potentials (SSEPs) prior to the tethered cord release. Conclusion Tethered cord surgery can be safely performed in patients after open fetal MMC closure without clinical decline in manual motor testing. Patients often have functional nerve roots below the anatomic level. Sensory function appears to be more severely affected in patients leading to a consistent motor-sensory imbalance. PubDate: 2022-11-16
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Abstract: Background The occipital interhemispheric transtentorial (OITT) approach is frequently used for accessing the pineal region. There are scarce reports of using the OITT to access superior cerebellar lesions. This approach affords the patient several advantages over traditional posterior fossa approaches. Purpose This study is to describe and evaluate clinical outcomes in a single surgeon case series of the OITT approach for pediatric patients with lesions of the superior cerebellum. Methods All pediatric patients who underwent an OITT craniotomy for a superior cerebellar lesion by a single surgeon over a 5-year period were included in this retrospective analysis. Patient demographics and clinical data were collected. Results Thirteen pediatric patients were identified. Cases included twelve tumors and one arteriovenous malformation. Gross total resection was achieved in 92% of cases. No patients developed posterior fossa syndrome. Two patients had transient homonymous hemianopsia that resolved by 1 month post-operatively. There were no permanent neurological deficits. Conclusion For superomedial cerebellar lesions presenting to the tentorial surface of the superior cerebellum in patients with normal to steep tentorial angles, the OITT approach is effective and safe. This approach has a low risk of posterior fossa syndrome and permanent visual deficits when applied appropriately. Patient selection is critical for maximizing the advantages of the OITT for superior cerebellar lesions. PubDate: 2022-11-16
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Abstract: Introduction Prompt detection of traumatic cervical spine injury is important as delayed or missed diagnosis can have disastrous consequences. Given the understood mechanism of non-accidental trauma (NAT), it is reasonable to suspect that cervical spine injury can occur. Current management of young children being evaluated for NAT includes placement of a rigid collar until clinical clearance or an MRI can be obtained. Currently, there exists a lack of robust data to guide cervical bracing. Anecdotally, our group has not observed a single patient with a diagnosis of NAT who required operative stabilization for cervical spine instability. This study will be the largest series to date and aims to systematically investigate this observation to determine the likelihood that children with a diagnosis of NAT harbor cervical spine instability related to their injuries. Methods Patient data from the Children’s Hospital Colorado Trauma Registry diagnosed with non-burn-only NAT were reviewed retrospectively. Children less than 4 years of age pulled from the registry from January 1, 2005, to March 31, 2021, were included. Demographic, admission/discharge, imaging, and clinic management data were collected for each patient and analyzed. Results There were 1008 patients included in the cohort. The age at presentation ranged from 5 days to 4 years (mean 10.4 months). No patient had X-ray or CT findings concerning for cervical instability. Three patients had MRI findings concerning for cervical instability. Two of these underwent external bracing, and the third died from unrelated injuries during their hospitalization. Only four patients were discharged in a cervical collar, and all were ultimately cleared from bracing. No patient underwent a spinal stabilization procedure. Conclusions While the mechanism of injury in many NAT cases would seem to make significant cervical spine injury possible, this single-center retrospective review of a large experience indicates that such injury is exceedingly rare. Further study is merited to understand the underlying pathophysiology. However, it is reasonable to consider cervical collar clearance in the setting of normal radiographs and a reassuring neurological exam. Furthermore, if concerns exist regarding cervical spine instability on MRI, an initial trial of conservative management is warranted. PubDate: 2022-11-15
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Abstract: Abstract This brief report is intended to call attention to the fact that we use some very old terms in our daily medical speaking that were in use about 3500 years ago and were probably uttered as early as the late Bronze Age by Achilles, Agamennon and the other Homeric heroes outside the walls of Troy. PubDate: 2022-11-12
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