Journal of Neurosurgery: Pediatrics
Volume 29: Issue 5 (May 2022)

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Masahiro Nonaka, Haruna Isozaki, Yumiko Komori, Takamasa Kamei, Junichi Takeda, Yuichiro Nonaka, Ichiro Yabe, Masayoshi Zaitsu, Kenji Nakashima, and Akio Asai

In Brief

To investigate how many surgeries patients with myelomeningocele have undergone in their lifetime, the number of surgeries listed in the claims of Japanese patients with myelomeningocele was tabulated. A total of 1033 surgeries were performed in 294 patients of various ages within a follow-up period of 4.4 years. A total of 0.42 surgeries were performed per patient per year. Patients with myelomeningocele underwent a large number of surgeries throughout their lives.

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Mandana Behbahani, Nathan A. Shlobin, Klaudia Dziugan, Hannah Callas, Theresa Meyer, Elizabeth Yerkes, Vineeta T. Swaroop, Michael DeCuypere, Sandi Lam, David McLone, and Robin Bowman

In Brief

In this study, researchers have investigated the long-term functional outcomes for a large cohort of patients born with a myelocystocele, with and without anomalies of cloacal development, treated at a single institution from 1982 to 2019. Patients with this complex form of closed spina bifida had favorable long-term outcomes in community ambulation and continence status. The results of this study have highlighted the importance of care by a multidisciplinary spina bifida team for this patient population.

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Paul A. Grabb, Emmanuel J. Vlastos, Paige A. Lundy, and Michael B. Partington

In Brief

The authors evaluated all infants with myelomeningocele for brainstem dysfunction to determine if prenatal closure had an impact. Prenatal closure of myelomeningocele was associated with a significant reduction in brainstem dysfunction. This should be an important factor to consider when counseling families considering prenatal closure of a myelomeningocele.

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Samuel J. Cler, Alexander Skidmore, Alexander T. Yahanda, Kimberly Mackey, Joshua B. Rubin, Andrew Cluster, Stephanie Perkins, Karen Gauvain, Allison A. King, David D. Limbrick Jr., Sean McEvoy, Tae Sung Park, Matthew D. Smyth, Ali Y. Mian, Michael R. Chicoine, Sonika Dahiya, and Jennifer M. Strahle

In Brief

The authors analyzed genetic and histopathological findings from a large cohort of pediatric pilocytic astrocytomas to identify predictors of tumor progression and recurrence. BRAF alteration status and histopathological findings were not associated with outcomes, although gross-total resection led to significantly lower rates of progression and recurrence. The prognostic value of genetic and histopathological findings in pediatric pilocytic astrocytoma is limited, and outcomes are primarily driven by extent of tumor resection.

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Stephano J. Chang, Ruth Mitchell, Juliette Hukin, and Ash Singhal

In Brief

Researchers performed a systematic review of cases of Holmes tremor secondary to hydrocephalus, to identify clinical associations, pathomechanisms, and treatment options. Obstructive triventriculomegaly with low pressure was a common association, as were oculomotor deficits, bradykinesia, and rigidity. CSF diversion and levodopa administration were the most common and effective treatments, providing insights into the pathophysiology of this rare and refractory condition.

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Victor M. Lu, Jessica D. Leuchter, Jamie E. Clarke, Evan M. Luther, Shelly Wang, and Toba N. Niazi

In Brief

Congenital heart disease and congenital cardiac anomalies may render endoscopic third ventriculostomy (ETV) and ventriculoperitoneal shunt (VPS) susceptible to failure in infants with hydrocephalus. Analysis of the Nationwide Inpatient Sample confirmed that congenital cardiac status can predict ETV and VPS failure in patients with infantile hydrocephalus. The authors hypothesized that this finding was primarily due to changes in central venous pressure but may not be completely universal across both interventions and all congenital cardiac anomalies.

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Katherine G. Holste, Clare M. Wieland, Mohannad Ibrahim, Hemant A. Parmar, Sara Saleh, Hugh J. L. Garton, and Cormac O. Maher

In Brief

In this study the authors aimed to define the prevalence of subdural hematomas (SDHs) and developmental outcomes in infants with benign expansion of the subarachnoid spaces (BESS). The prevalence of spontaneous SDH formation in the 480 included patients over a period of 25 years was 8.1%. There was no significant association between subarachnoid space size and SDH development. To the authors’ knowledge, this is the largest cohort of infants with BESS studied to date. Infants with BESS have a higher prevalence of spontaneous SDH formation than the general pediatric population.

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Jonathan Roth, Yurii Perekopaiko, Danil A. Kozyrev, Shlomi Constantini, and on behalf of the Pediatric Colloid Cyst Study Group (PCCSG)

In Brief

Pediatric colloid cysts (CCs) are rare. The goal of this study was to evaluate the clinical course of pediatric CCs. Pediatric CCs may present in all pediatric age groups, although most that are symptomatic present after the age of 10 years. There is a clear correlation between larger CCs and symptomatology, acuteness of symptoms, and need for surgery. Fifty percent of asymptomatic CCs progressed over time. The natural history of pediatric CCs is described in this largest series to date.

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Zahra Ebrahim Soltani, Sara Hanaei, Mohammad Amin Dabbagh Ohadi, Seyed Farzad Maroufi, Keyvan Tayebi Meybodi, Sima Khademi, Bahareh Yaghmaei, Alireza Ebrahim Soltani, Farideh Nejat, and Zohreh Habibi

In Brief

In this study the authors aimed to evaluate the safety and efficacy of tranexamic acid (TXA) versus aprotinin on the rate of blood loss and transfusion within the first 24 hours after craniosynostosis surgery. The adjusted intraoperative blood loss for weight (EBVloss) was lower with aprotinin administration, and both the 2- and 8-hour postoperative EBVloss values were significantly decreased when aprotinin or TXA was administered. These results demonstrated that aprotinin and TXA can reduce blood loss and transfusion without serious adverse events.

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Kaamya Varagur, Gary B. Skolnick, Sybill D. Naidoo, Matthew D. Smyth, and Kamlesh B. Patel

In Brief

This is one of the first long-term comparisons of outcomes following two primary repair types for unilateral coronal synostosis. Children who underwent open or endoscopic repair for this condition were found to have comparable facial symmetry at school age, but midface depth remained highly asymmetric in both groups. Forehead asymmetry at school age correlated with parent- and physician-reported desire for revision. Physical outcomes were comparable between groups, and further examination of psychosocial development in this population is warranted.

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Afshin Salehi, Peter H. Yang, and Matthew D. Smyth

In Brief

Invasive stereoelectroencephalography (SEEG) is increasingly utilized over subdural grid (SDG)—based techniques. However, the costs of these procedures are not well established, especially for pediatric patients. In a single-institution retrospective case series, the authors reported similar average total perioperative charges for SEEG ($106,291) and SDG ($104,442) when adjusted for inflation. This finding—along with the versatility, safety profile, and efficacy of SEEG—suggests that SEEG may be considered a first-line option for pediatric patients who require invasive monitoring.

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Joseph H. Garcia, Caleb Rutledge, Ethan A. Winkler, Luis Carrete, Ramin A. Morshed, Alex Y. Lu, Satvir Saggi, Christine K. Fox, Heather J. Fullerton, Helen Kim, Daniel L. Cooke, Steven W. Hetts, Michael T. Lawton, Nalin Gupta, and Adib A. Abla

In Brief

The objective of this study was to validate the recently created Ruptured Arteriovenous Malformation Grading Scale (RAGS) in a cohort of pediatric patients presenting with hemorrhagic brain arteriovenous malformations (AVMs), thereby demonstrating the scale's generalizability and expanding its external validity. The RAGS outperformed other clinical grading scales in predicting disability at three distinct follow-up periods. The RAGS score has the potential to become a much-needed tool for clinicians in the evaluation of patients, including children, presenting with hemorrhagic brain AVMs.

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William A. Lambert, Mitch Paro, Emanuela Pinci, Markus J. Bookland, Jonathan E. Martin, Rebecca Riba-Wolman, Laura McKay, and David S. Hersh

In Brief

Venous thromboembolism (VTE) is a rare but potentially devastating postoperative complication that appears to have a higher incidence among patients with central diabetes insipidus (DI). The authors conducted a systematic review of the literature to investigate the frequency of VTE in pediatric patients with central DI and report 2 additional patients treated at their institution. These findings suggest that pediatric neurosurgical patients with DI may benefit from more aggressive VTE surveillance and prophylaxis.

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Alexander P. Simko, Mark L. Hudak, Sabrina H. Han, Jinhai Huo, Kelsey Hayward, and Philipp R. Aldana

In Brief

The authors quantified the financial impact of a remote pediatric neurosurgery telemedicine clinic across state borders. Savings were substantial in comparison with an alternative in-person clinic and did not acutely impact care. Overall, the telemedicine clinic may serve as a model to those looking to set up a similar infrastructure for their patient populations.

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David S. Hersh, Jonathan E. Martin, and John C. Wellons III
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Ali Riazi, Firooz Salehpour, Farhad Mirzaei, and Ata Mahdkhah
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In Brief

The authors conducted a retrospective prognostic analysis aimed to elucidate the best treatment strategy among embryonal tumor with multilayered rosettes (ETMR) with confirmed chromosome 19 microRNA cluster (C19MC) amplification because most previously published reports included pathologically diagnosed cases regardless of C19MC status, for which treatment analysis may have been suboptimal because of molecular heterogeneity among the tumors. Important findings indicated that a multimodal treatment approach had a great impact on the prognosis of patients with C19MC-altered ETMR.

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The authors aimed to provide a cognitive, somatic, and radiological landscape of 100 consecutive children with a temporal arachnoid cyst discovered incidentally or not, but rarely for cognitive disorders. Half of children (50%) had no neuropsychological disorders, especially those with incidentally discovered cysts; the other half (50%) had selective cognitive and behavior disorders that may have a long-term impact on their daily life, particularly with a right-sided and/or convex cyst. Cyst size had little effect. This study may help in better selection of patients who may benefit from surgery.

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Researchers set out to study clinical, imaging, and neuropsychological changes in 34 selected children and teenagers with a temporal arachnoid cyst (TAC) 1 year after surgery. For more than half of children, TAC volume decreased = 50% and most somatic disorders disappeared; 76% of patients improved highly in IQs, language, memory, and executive functions, and children were less aggressive. When carefully selected (according to a decision-making algorithm), children with a TAC may benefit from surgery, regardless of age.

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Caregiver stress from a child's diagnosis can impact a caregiver's ability to participate in treatment decisions, comply, and manage long-term illness. The authors sought to compare caregiver stress in children with craniosynostosis at diagnosis and postoperatively. Stress levels ranged from normal to clinically significant in the caregivers, with sex, uninsured status, and open repair predicting higher stress. Stress decreased at 3 months postoperatively before increasing at 6 months. Intervention targeting caregiver stress should be explored to maintain lower stress observed at 3 months after surgery.

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The authors aimed, via systematic review and meta-analysis, to consolidate the literature surrounding endoscopic third ventriculostomy and choroid plexus cauterization (ETV+CPC) performance to better understand the trade-offs between the various hydrocephalus treatment options. The pooled estimate for the ETV+CPC failure rate was 44%, with a significant difference found between high- and lower-middle-income countries (53% vs 32%, p < 0.01). This study provides the most accurate estimates of ETV+CPC performance and highlights further research needs.

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Revascularization surgery for moyamoya is a specialized procedure for a rare condition that requires specific tissue-handling and microsurgical skills. In this paper, the authors demonstrate that in a high-volume center a newly appointed surgeon can be trained by an experienced one safely with no drop in quality and outcomes, and they also discuss the methods by which this can be achieved.

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In this study, the authors evaluated the differences in demographics, outcomes, and molecular features of brainstem, thalamic, and spinal cord gliomas harboring H3 K27M mutations. Brainstem lesions occur in younger patients, and these tumors are characterized by higher Ki-67 indices. Patients with brainstem tumors were demonstrated to have poorer survival in comparison to those with thalamic and spinal lesions. Genetically, H3 K27M–mutant diffuse midline gliomas in different anatomical locations also have distinct molecular profiles. This study demonstrated that H3 K27M–mutant diffuse midline gliomas have distinct characteristics, prognoses, and molecular profiles in different anatomical locations.

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In Brief

In this study, logistic regression models for each of the five concussion subtypes included predictors from multiple domains, highlighting the heterogeneous conceptualization of concussion and the importance of a multidomain assessment for this injury. The predictors identified in this study may guide a focused assessment and ultimately lead to more targeted interventions. Identifying clinical predictors that can differentiate concussion subtypes may inform a more efficient approach to concussion care that can reduce healthcare resource allocation and improve patient outcomes.

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In Brief

The goal of this study was to perform high-resolution 3D cranial bone MRI in pediatric patients without sedation. When compared with the gold standard (CT), the authors found that the 5-minute golden-angle stack-of-stars radial volumetric interpolated breath-hold examination MRI sequence was able to produce clinically acceptable cranial images with excellent ability to detect cranial vault fractures. This study will allow for future work to provide clinicians with a rapid diagnostic tool without radiation and sedation safety concerns.

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In Brief

The purpose of this study was to describe the safety and efficacy of endoscopic third ventriculostomy (ETV) revision after failure of initial ETV and choroid plexus cauterization (CPC). ETV revision had a significantly lower 1-year success rate than initial ETV+CPC and ventriculoperitoneal shunt (VPS) implantation. The question is what treatment to offer patients after failed initial ETV+CPC. ETV revision carries a 29.5% rate of success in the 1st year and similar complications to other hydrocephalus surgeries. We observed no change to the performance of subsequent VPS.

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The authors investigated radiological factors predictive of the extent of resection (EOR) in pediatric pineal region tumors through an occipital interhemispheric transtentorial approach (OITA). In 30 patients, a tumor volume < 2 cm3 above the Herophilus-Galen (H-G) plane, linear extension < 1 mm above the H-G line, and pineal histology were significantly predictive of gross-total resection achieved with the OITA. The amount of tumor extension above the H-G line of sight is a predictor of EOR for pineal tumor surgery through the OITA.

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In Brief

The Park-Reeves Syringomyelia Research Consortium database was used to compare complications and outcomes between posterior fossa decompression with and without duraplasty for Chiari I malformations. The authors found that posterior fossa decompression was associated with fewer complications and shorter hospital stays than posterior fossa decompression with duraplasty, although it was associated with a higher revision rate and lower rates of headache and syrinx improvement. This study, coupled with the forthcoming results of a randomized controlled trial, should help improve understanding regarding the indications for each surgery.

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This study aimed to quantitatively compare the effect of distraction osteogenesis (DO) on the cranial vault for the treatment of craniosynostosis according to patient age and distraction direction. Distraction in the first year of life demonstrated more balanced and greater expansion of the growing cranial vault in both the anterior and posterior DO groups. There were no significant differences in intracranial volume increases between the groups, indicating that both options are effective for cranial vault expansion. It is important when planning treatment to consider the notable difference in volumetric and morphological changes following DO according to the patient's age.

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In Brief

Prior studies have found that children with craniosynostosis may benefit from mental health care, rehabilitation therapies, and healthcare services supporting development. In this study, the authors quantified utilization of these services and found that more than 40% of children with craniosynostosis use such services, and that utilization does not differ between patients with syndromic and nonsyndromic cases. Although a multidisciplinary care approach is often used with syndromic patients, these findings suggest this model should be considered for nonsyndromic patients as well.

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To determine the demographic characteristics, presentation, management, and long-term outcome of intracranial aneurysms in infants ≤ 2 years of age, the authors performed an individual participant data meta-analysis of institutional case series data and a systematic review of infantile intracranial aneurysm cases reported in the contemporary literature. Most aneurysms were idiopathic, involved the anterior circulation, and presented with hemorrhage. Surgical and endovascular techniques yielded equally favorable outcomes, although surgery was favored for middle cerebral artery aneurysms and endovascular treatment for basilar aneurysms. Long-term outcomes compared favorably to those for adults, with 67% of infants recovering to a Glasgow Outcome Scale score of 5 at the latest follow-up.

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Transitioning from pediatric to adult care can be an anxious time for patients with hydrocephalus and their families. Using qualitative methodologies, researchers identified gaps in communication and a fear of what care would be like in an adult setting as significant concerns. The need for more independence was recognized and the importance of relationships with clinicians underscored. The results of this study can be used to design better transitioning models for hydrocephalus care.

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The use of endoscopic surgery for metopic synostosis is increasing, but objective outcome measures are lacking. In this pilot study of children undergoing endoscopic strip craniectomy with postoperative helmet orthosis therapy for metopic synostosis, the authors developed parameters to compare images obtained using 3D stereophotogrammetry, which avoids the use of anesthesia and ionizing radiation, in patients undergoing surgery for metopic synostosis and age-matched controls without synostosis. Analysis of changes in head shape after surgery showed that at 1 year postoperatively most metrics were the same for children in both groups.

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Researchers compared outcomes following open anterior cranial vault remodeling and endoscopic strip craniectomy (ESC) for patients with Apert syndrome and bilateral coronal craniosynostosis. Their research demonstrated that patients with Apert syndrome treated with ESC had head growth that was not inferior to that of patients treated with open cranial vault remodeling as well as to age- and sex-matched peers. Furthermore, the minimally invasive ESC procedure demonstrated statistically significantly shorter length of hospital stay, shorter surgical time, and less blood loss.

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The authors describe a patient with mesial temporal sclerosis who had undergone laser interstitial thermal therapy (LITT) at a separate institution with no post-LITT improvement and who then subsequently attained seizure freedom after stereotactic electroencephalography—guided anterior temporal lobectomy. They emphasize the surgical adjuncts and technical considerations helpful to achieve safe post-LITT resection of mesial temporal structures.

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Researchers investigated the proportion of favorable outcomes for epileptic spasm (ES) by disconnection surgery, the preoperative factors related to these favorable outcomes, and the timing of postoperative ES recurrence. A total of 75.6% of patients with ES achieved favorable outcomes. Longer seizure duration, presence of hypsarrhythmia, and positive MRI findings led to poorer postoperative outcomes. The predominant period of ES recurrence was less than 13 months. Disconnection surgery is an important treatment option for medically intractable ES.

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Differences between outcome measures that are clinician-dependent versus those reflecting real-world movements remain unexplored. Technological advances, such as accelerometry, can address that knowledge gap. The authors established the feasibility of such technology in a pediatric population after nerve reconstruction, and demonstrate that postsurgical patients use their reconstructed arms 80% of the time they use their unaffected arms. These data represent the first long-term, real-world evidence to support nerve reconstruction for patients affected by neonatal brachial plexus palsies.

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The authors describe their initial experience with MRI-guided laser interstitial thermal therapy (MRgLITT) assisted by a robotic arm for the treatment of hypothalamic hamartomas in pediatric patients with refractory epilepsy. Sixteen procedures were performed in 11 patients. The robotic arm was found to be a precise and safe tool to implant the laser fiber and MRgLITT an effective technique to treat these patients. Complications were also analyzed. This is the first European series of MRgLITT in epilepsy surgery and in pediatric patients.

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Surgeons in the Hydrocephalus Clinical Research Network implemented a simplified infection prevention protocol for shunt surgery. Compliance was 79.4% and nonprotocol factors were recorded. In 4913 procedures, the infection rate was 5.1%. The presence of = 2 complex chronic conditions and recent shunt surgery (within 12 weeks) were independent predictors of infection. The use of antibiotic-impregnated catheters and vancomycin irrigation were independent factors protective against shunt infection.

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Given the paucity of data on the clinical behavior of brainstem cavernous malformations (BSCMs) in childhood and on the long-term outcome in children managed conservatively and surgically, the experiences from 2 major European centers were reviewed. Surgical treatment should be considered early in young patients presenting with surgically accessible lesions and an aggressive clinical course, and it should be performed in a high-volume center. This study further defines the role of surgery in the management of children with BSCMs.

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Meta-analysis was used to examine the literature on the effectiveness of vagus nerve stimulation (VNS) in children with reported genetic etiologies of drug-resistant epilepsy. Tuberous sclerosis complex patients had substantial seizure reduction and improvements in quality of life. Dravet syndrome patients had less robust seizure reduction and variable improvements in cognition. Limited evidence exists regarding VNS outcomes in patients with other known genetic etiologies. This study highlights the increasing importance of genetic diagnosis. Long-term follow-up is needed.

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Researchers used an electrical circuit model of pulsatility in the cranium to explain why ventricles dilate in obstructive hydrocephalus but not in pseudotumor cerebri. They found that ventriculomegaly can be understood as an adaptation to impairment of the windkessel mechanism in obstructive hydrocephalus, but the windkessel works properly in pseudotumor. This finding provides insight into the pathophysiology of pseudotumor and obstructive hydrocephalus and may help to design more effective treatments.

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The authors present their experience in awake brain surgery and assess the value of using electrocorticography to detect afterdischarge potentials (ADP). The interpretation of the results from language mapping during the awake phase may be misguided by the occurrence of clinically relevant ADP. Clinicians should be aware of this finding, and monitoring of ADP in awake brain surgery should be a standard.

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In Brief

Researchers developed a novel method to quantify the complex head shape and frontal retrusion in metopic craniosynostosis. The presented method objectively quantified complex head shape and both frontal and supraorbital retrusion while avoiding radiation exposure in patients with metopic craniosynostosis. It may also provide a quantitative measure for follow-up after surgical treatment using endoscopic suturectomy.

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This study elucidated the physiological growth of spinal lipomas in the early postnatal period. The rapid growth of spinal lipomas was observed in patients younger than 3 months, especially in those younger than 1 month, in pace with growth of the normal subcutaneous fat and increase in BMI. These findings support the importance of close clinical observation and careful planning for surgery in this period.

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The objective of this study was to assess for demographic and socioeconomic disparities among patients treated with fetal or postnatal myelomeningocele (MMC) closure. The authors found that the study population was not representative of the state's population with MMC when considering race and insurance status despite patients traveling from across the state for care. These findings suggest upstream disparities in access to evaluation for fetal surgery and highlight the need to consider access as part of a new treatment's success.

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In this systematic review and individual patient data meta-analysis assessing the outcomes of spinal column shortening (SCS) for the treatment of tethered cord syndrome (TCS), it was found that SCS is safe and effective in relieving preoperative symptoms of pain, weakness, and bowel and bladder dysfunction (level C evidence; class IIb recommendation). This study further illuminates the utility of SCS and the need for prospective comparative studies of SCS and traditional tethered cord release for TCS.

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In a large cohort of children with severe traumatic brain injury undergoing emergency hemicraniectomy, researchers identified diffuse axonal injury in the diencephalon and brainstem as powerful predictors of poor outcome.

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Social determinants of health can have a profound effect on outcomes. The authors sought to determine if there are sociodemographic differences between patients who underwent fetal surgery versus those who underwent postnatal surgery for myelomeningocele. There was a significant difference in the commercial insurance rate and a marked difference in the racial and ethnic makeup of the two groups. Future studies should account for these demographic differences, and fetal centers should commit to minimizing preventable disparities.

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In this systematic review, the authors sought to review the published evidence on the use of cranial neuromodulation—deep brain stimulation (DBS) and responsive neurostimulation (RNS)—for pediatric drug-resistant epilepsy (DRE). From 35 studies, they found 72 children who had undergone DBS and 42 who had undergone RNS. Overall, 75% and 73.2% of DBS and RNS patients, respectively, experienced > 50% reduction in seizures. This study shows the paucity of evidence for promising treatment options for pediatric DRE.

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Investigators applied lesion network mapping to children with focal cortical dysplasia who underwent surgery for drug-resistant epilepsy. Lesion volumes with persistent seizures after surgery tended to have stronger connectivity to attention and motor networks and weaker connectivity to the default mode network, compared with lesion volumes with seizure-free surgical outcomes. Network connectivity–based lesion-outcomes mapping may offer new insight for determining the impact of lesion volumes discerned according to both size and specific location.

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The goal of this paper was to describe a neurosurgical educational partnership and the early results in developing pediatric craniofacial expertise in Southeast Asia. After brief, focused training through partnership with an institution in the United States followed by consistent telecollaboration, Vietnamese neurosurgical colleagues were able to rapidly scale up treatment of craniosynostosis with good outcomes. Global neurosurgical partnerships are feasible and effective methods of education and development of local neurosurgical expertise in low- and middle-income countries.