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Open access

Lance M. Villeneuve, Murali Nagarajan, Benjamin O. Cornwell, Laura Propester, Xiaochun Zhao, Jami Valenzuela, Joanna Gernsback, Virendra Desai, and Andrew Jea

BACKGROUND

“Torcular pseudomass,” or redundant soft tissue in the torcular region, is not an infrequent incidental finding on advanced imaging of the brain in infants and young children. It was recently codified among pediatric neuroradiologists; however, its report in the pediatric neurosurgical community has not previously been elucidated.

OBSERVATIONS

The authors present a case of a 14-month-old child who presented with fever and a first-time seizure. Computed tomography of the head suggested an epidural abscess; however, magnetic resonance imaging characteristics of the lesion were consistent with torcular pseudomass, a normal variant. At the 3-month follow-up, the child was continuing to do well and had not had another seizure. There have been no indications for surgical intervention or additional radiographic surveillance.

LESSONS

The differential diagnosis for torcular pseudomass includes dural venous sinus thrombosis, dermoid cysts, occipital encephalocele, eosinophilic granuloma, and primary and metastatic tumors, such as neuroblastoma. The management of each of these disorders in the differential diagnosis may be much more invasive than continued observation in the case of torcular pseudomass. Therefore, it is important for pediatric neurosurgeons to become familiar with this developmental anomaly of the dura and occipital skull.

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*Yosef M. Dastagirzada, Nikita G. Alexiades, David B. Kurland, Sebastián N. Anderson, Douglas L. Brockmeyer, David B. Bumpass, Sandip Chatterjee, Mari L. Groves, Todd C. Hankinson, David Harter, Daniel Hedequist, Andrew Jea, Jeffrey R. Leonard, Jonathan E. Martin, Matthew E. Oetgen, Joshua Pahys, Curtis Rozzelle, Jennifer M. Strahle, Dominic Thompson, Burt Yaszay, and Richard C. E. Anderson

OBJECTIVE

Cervical spine disorders in children are relatively uncommon; therefore, paradigms for surgical and nonsurgical clinical management are not well established. The purpose of this study was to bring together an international, multidisciplinary group of pediatric cervical spine experts to build consensus via a modified Delphi approach regarding the clinical management of children with cervical spine disorders and those undergoing cervical spine stabilization surgery.

METHODS

A modified Delphi method was used to identify consensus statements for the management of children with cervical spine disorders requiring stabilization. A survey of current practices, supplemented by a literature review, was electronically distributed to 17 neurosurgeons and orthopedic surgeons experienced with the clinical management of pediatric cervical spine disorders. Subsequently, 52 summary statements were formulated and distributed to the group. Statements that reached near consensus or that were of particular interest were then discussed during an in-person meeting to attain further consensus. Consensus was defined as ≥ 80% agreement on a 4-point Likert scale (strongly agree, agree, disagree, strongly disagree).

RESULTS

Forty-five consensus-driven statements were identified, with all participants willing to incorporate them into their practice. For children with cervical spine disorders and/or stabilization, consensus statements were divided into the following categories: A) preoperative planning (12 statements); B) radiographic thresholds of instability (4); C) intraoperative/perioperative management (15); D) postoperative care (11); and E) nonoperative management (3). Several important statements reaching consensus included the following recommendations: 1) to obtain pre-positioning baseline signals with intraoperative neuromonitoring; 2) to use rigid instrumentation when technically feasible; 3) to provide postoperative external immobilization for 6–12 weeks with a rigid cervical collar rather than halo vest immobilization; and 4) to continue clinical postoperative follow-up at least until anatomical cervical spine maturity was reached. In addition, preoperative radiographic thresholds for instability that reached consensus included the following: 1) translational motion ≥ 5 mm at C1–2 (excluding patients with Down syndrome) or ≥ 4 mm in the subaxial spine; 2) dynamic angulation in the subaxial spine ≥ 10°; and 3) abnormal motion and T2 signal change on MRI seen at the same level.

CONCLUSIONS

In this study, the authors have demonstrated that a multidisciplinary, international group of pediatric cervical spine experts was able to reach consensus on 45 statements regarding the management of pediatric cervical spine disorders and stabilization. Further study is required to determine if implementation of these practices can lead to reduced complications and improved outcomes for children.

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Virendra R. Desai, Audrey Grossen, Huy Gia Vuong, Nicholas Hopkins, Mikayla Peters, and Andrew Jea

OBJECTIVE

COVID-19 has not only impacted healthcare systems directly via hospitalizations and resource utilization, but also indirectly via adaptations in healthcare practice, such as the evolution of the academic environment and the rise of telemedicine and virtual education. This void in clinical responsibilities has been filled with academic productivity in various fields. In this study the authors investigate the influence of COVID-19 on the academic focus within pediatric neurosurgery.

METHODS

All data were obtained from the Journal of Neurosurgery: Pediatrics (JNS Peds). The number of submissions for each month from January 2017 to December 2021 was collected. Data including number of publications, publication level of evidence (LOE), and COVID-19–related articles were collected and verified. Each publication was categorized by manuscript and LOE according to adaptations from the Canadian Task Force on Periodic Health Examination. Publication groups were categorized as pre–COVID-19 (January 2017–February 2020), peri–COVID-19 (March 2020–July 2020), and post–COVID-19 (August 2020–December 2021). Statistical analysis was performed to compare pre–COVID-19, peri–COVID-19, and post–COVID-19 academic volume and quality.

RESULTS

During the study time period, a total of 3116 submissions and 997 publications were identified for JNS Peds. Only 2 articles specifically related to COVID-19 and its impact on pediatric neurosurgery were identified, both published in 2021. When analyzing submission volume, a statistically significant increase was seen during the shutdown relative to pre–COVID-19 and post-shutdown time periods, and a significant decrease was seen post-shutdown relative to pre–COVID-19. LOE changed significantly as well. When comparing pre–COVID-19 versus post–COVID-19 articles, a statistically significant increase was identified only in level 4 publications. When analyzing pre–COVID-19 versus post–COVID-19 (2020) and post–COVID-19 (2021), a statistically significant decrease in level 3 and increases in levels 4 and 5 were identified during post–COVID-19 (2020), with a rebound increase in level 3 and a decrease in level 5 during post–COVID-19 (2021).

CONCLUSIONS

There was a significant increase in manuscript submission during the initial pandemic period. However, there was no change during subsequent spikes in COVID-19–related hospitalizations. Coincident with the initial surge in academic productivity, despite steady publication volume, was an inverse decline in quality as assessed by LOE.

Free access

Audrey Grossen, Theresa Gavula, Deepti Chrusciel, Alexander Evans, Rene McNall-Knapp, Ashley Taylor, Benay Fossey, Margaret Brakefield, Carrick Carter, Nadine Schwartz, Naina Gross, Andrew Jea, and Virendra Desai

OBJECTIVE

Neurocutaneous syndromes have variable multisystem involvement. The multiorgan involvement, potential pathologies, and various treatment options necessitate collaboration and open discussion to ensure optimal treatment in any given patient. These disorders provide quintessential examples of chronic medical conditions that require a lifelong, multidisciplinary approach. The objectives of this study were to 1) perform a systematic review, thoroughly assessing different multidisciplinary clinic layouts utilized in centers worldwide; and 2) characterize an institutional experience with the management of these conditions, focusing on the patient demographics, clinical presentation, complications, and therapeutic strategies seen in a patient population.

METHODS

A systematic review of studies involving multidisciplinary clinics and their reported structure was performed according to PRISMA guidelines using the PubMed database. Then a retrospective chart review of patients enrolled in the Oklahoma Children’s Hospital Neurocutaneous Syndromes Clinic was conducted.

RESULTS

A search of the PubMed database yielded 251 unique results. Of these, 15 papers were included in the analysis, which identified 16 clinics that treated more than 2000 patients worldwide. The majority of these clinics treated patients with neurofibromatosis (13/16). The remaining clinics treated patients with von Hippel–Lindau syndrome (n = 1), tuberous sclerosis complex (n = 1), and multiple neurocutaneous syndromes (n = 1). The most commonly represented subspecialties in these clinics were genetics (15/16) and neurology (13/16). Five clinics (31%) solely saw pediatric patients, 10 clinics saw a combination of children and adults, and the final clinic had separate pediatric and adult clinics. The retrospective chart review of the Neurocutaneous Syndromes Clinic demonstrated that 164 patients were enrolled and seen in the clinic from April 2013 to December 2021. Diagnoses were made based on clinical findings or results of genetic testing; 115 (70%) had neurofibromatosis type 1, 9 (5.5%) had neurofibromatosis type 2, 35 (21%) had tuberous sclerosis complex, 2 (1%) had von Hippel–Lindau syndrome, 2 (1%) had Gorlin syndrome, and the remaining patient (0.6%) had Aarskog-Scott syndrome. Patient demographics, clinical presentation, complications, and therapeutic strategies are also discussed.

CONCLUSIONS

To the best of the authors’ knowledge, this is the first detailed description of a comprehensive pediatric neurocutaneous clinic in the US that serves patients with multiple syndromes. There is currently heterogeneity between described multidisciplinary clinic structures and practices. More detailed accounts of clinic compositions and practices along with patient data and outcomes are needed in order to establish the most comprehensive and efficient multidisciplinary approach for neurocutaneous syndromes.

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Huy Gia Vuong, Hieu Trong Le, Andrew Jea, Rene McNall-Knapp, and Ian F. Dunn

OBJECTIVE

The prognostic significance and genetic characteristics of H3 K27M–mutant diffuse midline gliomas (DMGs) in different anatomical locations requires further clarification. In this study, the authors integrated published data to investigate the differences between brainstem, thalamic, and spinal cord tumors.

METHODS

PubMed and Web of Science databases were used to search for eligible articles. Studies were included if they provided individual patient data of H3 K27M–mutant DMGs with available tumor locations. Hazard ratios (HRs) and 95% confidence intervals (CIs) were computed to investigate the survival of each subgroup.

RESULTS

Eight hundred four tumors were identified, including 467, 228, and 109 in the brainstem, thalamus, and spine, respectively. Brainstem tumors were primarily observed in young children, while patients with thalamic and spinal cord tumors afflicted older patients. The Ki-67 labeling index was highest in brainstem tumors. Compared to patients with brainstem tumors, those with thalamic (HR 0.573, 95% CI 0.463–0.709; p < 0.001) and spinal cord lesions (HR 0.460, 95% CI 0.341–0.621; p < 0.001) had a significantly better survival. When patients were stratified by age groups, superior overall survival (OS) of thalamic tumors was observed in comparison to brainstem tumors in young children and adolescents, whereas adult tumors had uniform OS regardless of anatomical sites. Genetically, mutations in HIST1H3B/C (H3.1) and ACVR1 genes were mostly detected in brainstem tumors, whereas spinal cord tumors were characterized by a higher incidence of mutations in the TERT promoter.

CONCLUSIONS

This study demonstrated that H3 K27M–mutant DMGs have distinct clinical characteristics, prognoses, and molecular profiles in different anatomical locations.

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Luke G. McVeigh, Miracle C. Anokwute, Sixia Chen, and Andrew Jea

OBJECTIVE

Tethered cord release (TCR) is the gold standard treatment for tethered cord syndrome (TCS); however, there are significant shortcomings including high rates of retethering, especially in complex and recurrent cases. Spinal column shortening (SCS) is an alternative treatment for TCS intended to avoid these shortcomings. Early studies were limited to case reports and smaller case series; however, in recent years, larger case series and small cohort studies have been conducted. Given the increase in available data, a repeat systematic review and meta-analysis is warranted to assess the safety and efficacy of SCS for TCS.

METHODS

The authors conducted a systematic review using MEDLINE (OVID), Embase (Elsevier), and Web of Science records dating from 1944 to July 2021 to identify all articles investigating SCS for TCS. They performed standard and individual patient data (IPD) meta-analyses, with 2 independent reviewers using PRISMA-IPD guidelines. Primary outcomes were improvement of preoperative clinical symptoms of pain, motor weakness, and bladder and bowel dysfunction, and also surgical complication rate. Secondary outcomes included urodynamic improvement and health-related quality-of-life outcomes determined using patient-reported outcome tools. Individual study quality assessment was performed using a standardized assessment tool for case reports/series, and publication bias was assessed using funnel plot analyses.

RESULTS

The review yielded 15 studies with 191 cases of TCS treated with SCS. IPD were available in 11 studies with 89 cases. The average age at time of surgery was 28.0 years (range 5–76 years). The average follow-up time was 33.2 months (range 7–132 months). Improvement was observed at last follow-up in 60 of 70 (85.7%) patients with preoperative pain, in 38 of 60 (60.3%) patients with preoperative weakness, and in 36 of 76 (47.4%) patients with preoperative bladder or bowel dysfunction. Complications of CSF leak, new neurological deficit, wound infection, or reoperation occurred in 4 of 89 (4.5%) patients.

CONCLUSIONS

SCS may be considered a safe and efficacious treatment option for TCS in children and adults (level C evidence; class IIb recommendation), especially for recurrent and complex cases. Current evidence is likely to be affected by selection and publication bias. Prospective comparative studies of SCS and TCR for TCS are recommended to determine long-term duration of outcomes, long-term safety in skeletally immature children, and exact indications of SCS versus traditional TCR.

Free access

Laura G. Valentini and Ignazio G. Vetrano

Free access

Rajiv R. Iyer and Douglas L. Brockmeyer