Global pediatric craniopharyngioma management modalities and outcomes.

INTRODUCTION: Pediatric craniopharyngioma is a complex pathology, with optimal management involving a multidisciplinary approach and thoughtful care coordination. To date, no studies have compared various treatment modalities and outcomes described in different global regions. We conducted a comprehensive systematic review to compare demographics, clinical presentation, treatment approach and outcomes of children diagnosed with craniopharyngioma globally. METHODS: A systematic review was conducted in accordance with the Preferred Reporting Item for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Search terms included "craniopharyngioma" and country-specific terms. Inclusion criteria included full-text studies published between 2000-2022, primarily examining pediatric patients 18-years old or younger diagnosed with craniopharyngioma, and reporting management and outcomes of interest. Data extracted included country of origin, demographical data, initial presentation and treatment modality, and outcomes. Descriptive statistics and between-group comparisons based on country of origin were performed. RESULTS: Of 797 search results, 35 articles were included, mostly originating from high-income countries (HIC) (n = 25, 71.4%). No studies originated from low-income countries (LIC). When comparing HIC to middle-income countries (MIC), no differences in patient demographics were observed. No differences in symptomatology at initial presentation, tumor type, surgical approach or extent of surgical resection were observed. HIC patients undergoing intracystic therapy were more likely to receive bleomycin (n = 48, 85.7%), while the majority of MIC patients received interferon therapy (n = 10, 62.5%). All MIC patients undergoing radiation therapy underwent photon therapy (n = 102). No statistically significant differences were observed in postoperative complications or mean follow-up duration between HIC and MIC (78.1 ± 32.2 vs. 58.5 ± 32.1 months, p = 0.241). CONCLUSION: Pediatric craniopharyngioma presents and is managed similarly across the globe. However, no studies originating from LICs and resource-poor regions examine presentation and management to date, representing a significant knowledge gap that must be addressed to complete the global picture of pediatric craniopharyngioma burden and management.

Pediatric neurosurgical-oncology scope and management paradigms in Sub-Saharan Africa: a collaboration among 7 referral hospitals on the subcontinent.

Background: Understanding of the epidemiology and biology of pediatric CNS tumors has advanced dramatically over the last decade; however there remains a discrepancy in the understanding of epidemiologic data and clinical capacity between high- and lower-income countries. Objective: We collected and analyzed hospital-level burden and capacity-oriented data from pediatric neurosurgical oncology units at 7 referral hospitals in Sub-Saharan Africa (SSA). Methods: A cross sectional epidemiological survey was conducted using REDCap at the 7 SSA sites, capturing 3-month aggregate data for patients managed over a total of 9 months. Descriptive statistical analyses for the aggregate data were performed. Results: Across the neurosurgical spectrum, 15% of neurosurgery outpatient and 16% of neurosurgery operative volume was represented by pediatric neuro-oncology across the 7 study sites. Eighty-six percent and 87% of patients who received surgery underwent preoperative CT scan and/or MRI respectively. Among 312 patients evaluated with a CNS tumor, 211 (68%) underwent surgery. Mean surgery wait time was 26.6 ± 36.3 days after initial presentation at the clinic. The most common tumor location was posterior fossa (n=94, 30%), followed by sellar/suprasellar region (n=56, 18%). Histopathologic analysis was performed for 189 patients (89%). The most common pathologic diagnosis was low grade glioma (n=43, 23%), followed by medulloblastoma (n=37, 20%), and craniopharyngioma (n=31, 17%). Among patients for whom adjuvant therapy was indicated, only 26% received chemotherapy and 15% received radiotherapy. Conclusion: The histopathologic variety of pediatric brain and spinal tumors managed across 7 SSA referral hospitals was similar to published accounts from other parts of the world. About two-thirds of patients received a tumor-directed surgery with significant inter-institutional variability. Less than a third of patients received adjuvant therapy when indicated. Multi-dimensional capacity building efforts in neuro-oncology are necessary to approach parity in the management of children with brain and spinal tumors in SSA.

Global Epidemiology of Pediatric Traumatic Spine Injury: A Systematic Review and Meta-Analysis.

OBJECTIVE: Traumatic spine injury (TSI) leads to significant morbidity and mortality in children. However, the global epidemiology of pediatric TSI is currently unknown. We conducted a systematic review and meta-analysis to estimate the global incidence of pediatric TSI and the burden of cases. METHODS: PubMed, Embase, and Scopus were searched for reports in June 2021 and updated in March 2023 with no restrictions on language or year of publication. A meta-analysis was conducted to estimate the global incidence of pediatric TSI and, subsequently, the number of cases of pediatric TSI worldwide and the proportion requiring spine surgery. RESULTS: Of 6557 studies, 25 met the inclusion criteria. Road traffic accidents (64%) were responsible for most cases reported in the literature, followed by falls (18%). The global incidence of TSI in children aged ≤20 years was estimated to be 14.24 of 100,000 children, or 375,734 children, with an estimated 114,975 requiring spine surgery. Across the World Bank income classification groups, lower middle-income countries had the highest pediatric TSI case burden (186,886 cases, with 57,187 requiring spine surgery). Across the World Health Organization regions, countries in the Southeast Asia region had the largest number of projected cases at 88,566, with 27,101 requiring surgical management, followed closely by the African region, with 87,235 projected cases and 26,694 requiring surgical management. CONCLUSIONS: Pediatric TSI represents a large healthcare burden globally. Interventions targeting both injury prevention and strengthening of neurosurgical capacity, especially in low resource settings, are needed to address this global health challenge.

A Comparison of Surgery Wait Times and Postoperative Length of Hospital Stay Among Patients with Brain Tumors by Country-Level Income and Healthcare System: A Systematic Review and Meta-analysis.

OBJECTIVE: The current study highlights the differences in surgery wait times and postoperative length of hospital stay (LOS) for brain tumor patients between high income countries (HICs) and low- and middle-income countries (LMICs), and across countries with different payer health systems. METHODS: A systematic review and meta-analysis were performed in accordance with the Preferred Reporting Items of Systematic Reviews and Meta-analyses (PRISMA) guidelines. Outcomes of interest were surgery wait time and postoperative LOS. RESULTS: Fifty-three articles were included totaling 456,432 patients. Five studies discussed surgery wait times and 27 discussed LOS. Three HIC studies reported mean surgery wait time of 4 days (SD not reported), 33 ± 13 days, and 34 ± 39 days, and 2 LMIC studies reported median surgery wait time of 4.6 (1-15) and 50 (13-703) days. Mean LOS was 5.1 days (95% CI: 4.2-6.1 days) from 24 HIC studies and 10.0 days (95% CI: 4.6-15.6 days) from 8 LMIC studies respectively. Mean LOS was 5.0 days (95% CI: 3.9-6.0 days) from countries with mixed payer system, and 7.7 days (95% CI: 4.8-10.5 days) from countries with single payer systems. CONCLUSIONS: There are limited data on surgery wait-times yet slightly more data on postoperative LOS. Despite a wide range of wait times, mean LOS in brain tumor patients tended to be longer in LMICs than HICs and longer for countries with single payer health systems than mixed payer health systems. Further studies are needed to evaluate surgery wait times and LOS for brain tumor patients more accurately.

Treatment of hydrocephalus following posterior fossa tumor resection: a multicenter collaboration from the Hydrocephalus Clinical Research Network.

OBJECTIVE: Persistent hydrocephalus following posterior fossa brain tumor (PFBT) resection is a common cause of morbidity in pediatric brain tumor patients, for which the optimal treatment is debated. The purpose of this study was to compare treatment outcomes between VPS and ETV in patients with persistent hydrocephalus following surgical resection of a PFBT. METHODS: A post-hoc analysis was performed of the Hydrocephalus Clinical Research Network (HCRN) prospective observational study evaluating VPS and ETV for pediatric patients. Children who experienced hydrocephalus secondary to PFBT from 2008 to 2021 were included. Primary outcomes were VPS/ETV treatment failure and time-to-failure (TTF). RESULTS: Among 241 patients, the VPS (183) and ETV (58) groups were similar in age, extent of tumor resection, and preoperative ETV Success Score. There was no difference in overall treatment failure between VPS and ETV (33.9% vs 31.0%, p = 0.751). However, mean TTF was shorter for ETV than VPS (0.45 years vs 1.30 years, p = 0.001). While major complication profiles were similar, compared to VPS, ETV patients had relatively higher incidence of minor CSF leak (10.3% vs. 1.1%, p = 0.003) and pseudomeningocele (12.1% vs 3.3%, p = 0.02). No ETV failures were identified beyond 3 years, while shunt failures occurred beyond 5 years. Shunt infections occurred in 5.5% of the VPS cohort. CONCLUSIONS: ETV and VPS offer similar overall success rates for PFBT-related postoperative hydrocephalus. ETV failure occurs earlier, while susceptibility to VPS failure persists beyond 5 years. Tumor histology and grade may be considered when selecting the optimal means of CSF diversion.

Understanding the Ghanaian Neurosurgical Literature: A Scoping Review and Bibliometric Analysis.

BACKGROUND: Research serves to bolster clinical neurosurgery by critically assessing various disease pathologies, while identifying important challenges and opportunities. However, there is limited information on the landscape of the Ghanaian neurosurgical literature. METHODS: A scoping review and bibliometric analysis was conducted in accordance with PRISMA guidelines. PubMed, Embase, Global Index Medicus, and Web of Science electronic databases were searched from inception until December 21, 2021 for English language articles about neurosurgery in Ghana. RESULTS: 927 articles were identified and 66 were ultimately included in the analysis. A majority of them, 42.4%, were retrospective cohort studies, with 62.1% published after 2010. There were no randomized controlled or basic science studies. Most articles were published in the West African Journal of Medicine (24.2%) and non-infectious/non-traumatic spinal pathology was the most commonly discussed topic (22.7%); 66.7% of articles included only authors affiliated with Ghanaian institutions, and international collaborators frequently originated from the United States (15.9%). Only 22.7% of the manuscripts reported a funding source. Commonly reported challenges included limited sample sizes, delays in diagnosis and treatment, and lack of proper diagnostic tools and specialized care. CONCLUSIONS: This review revealed that while the Ghanaian academic neurosurgery output has been increasing over time, these have been limited to cohort studies largely assessing spine pathology. The Ghanaian neurosurgical research environment may be bolstered by an increase in research funding, the establishment of longitudinal clinical databases, training in research methodology, increased incentives for researchers, strengthening of research collaborative networks, and increased engagement of neurosurgical trainees in research.

Clinical and molecular features of disseminated pediatric low-grade glioma and glioneuronal tumors: a systematic review and survival analysis.

Background: Disseminated pediatric low-grade gliomas and glioneuronal tumors (dpLGG/GNTs) are associated with a poorer prognosis than nondisseminated pLGG/GNTs. To date there is no comprehensive report characterizing the genome profile of dpLGG/GNTs and their relative survival. This systematic review aims to identify the pattern of genetic alterations and long-term outcomes described for dpLGG/GNT. Methods: A systematic review of the literature was performed to identify relevant articles. A quality and risk of bias assessment of articles was done using the GRADE framework and ROBINS-I tool, respectively. Results: Fifty studies published from 1994 to 2020 were included in this review with 366 cases reported. There was sporadic reporting of genetic alterations. The most common molecular alterations observed among subjects were 1p deletion (75%) and BRAF-KIAA1549 fusion (55%). BRAF p.V600E mutation was found in 7% of subjects. A higher proportion of subjects demonstrated primary dissemination compared to secondary dissemination (65% vs 25%). First-line chemotherapy consisted of an alkylation-based regimen and vinca alkaloids. Surgical intervention ranged from biopsy alone (59%) to surgical resection (41%) and CSF diversion (28%). Overall, 73% of cases were alive at last follow-up. Survival did not vary by tumor type or timing of dissemination. All studies reviewed either ranked low or moderate for both quality and risk of bias assessments. Conclusions: Chromosome 1p deletion and BRAF-KIAA1549 fusion were the most common alterations identified in dpLGG/GNT cases reviewed. The relative molecular heterogeneity between DLGG and DLGNT, however, deserves further exploration and ultimately correlation with their biologic behavior to better understand the pathogenesis of dpLGG/GNT.

Assessment of the Neurosurgical Capacity in Ghana: Challenges and Opportunities.

BACKGROUND: Low- and middle-income countries experience numerous challenges in the provision of neurosurgical care. However, limited information exists on the neurosurgical workforce and the constraints under which care is delivered in Ghana, West Africa. METHODS: A 19-item survey assessing neurosurgical workforce, infrastructure, and education was administered to Ghanaian consultant neurosurgeons and neurosurgeon trainees between November 8, 2021, and January 20, 2022. The data were analyzed using summary descriptions, and qualitative data were categorized into themes. RESULTS: There were 25 consultant neurosurgeons and 8 neurosurgical trainees (from 2 training centers) identified at 11 hospitals in Ghana totaling a workforce density of 1 neurosurgeon per 1,240,000. Most neurosurgical centers were located in Accra, the capital city. Almost half of the population did not have access to a hospital with a neurosurgeon in their region. Of hospitals, 82% had in-house computed tomography and/or magnetic resonance imaging scanners. In the operating room, most neurosurgeons had access to a high-speed drill (91%) but lacked microscopes and endoscopic sets (only 64% and 36% had these tools, respectively). There were no neurointensivists or neurological intensive care units in the entire country, and there was a paucity of neurovascular surgeries and functional neurosurgical procedures. CONCLUSIONS: The provision of neurosurgical care in Ghana has come a long way since the 1960s. However, the neurosurgical community continues to face significant challenges. Alleviating these barriers to care will call for systems-level changes that allow for the prioritization of neurosurgical care within the Ghanaian health care system.

Barriers Encountered Toward Pursuing a Neurosurgical Career: A Cross-Sectional Study Among Medical Students, Interns, and Junior Doctors in Africa.

BACKGROUND: Africa has an increased burden of neurosurgical diseases with an estimate of 1,986,392 neurosurgical cases, 108,824 neurosurgical case capacity, and 1,877,568 case deficits yearly. Literature suggests that about 8420 neurosurgeons are needed to fill this vast gap. The main objective of this study is to elucidate barriers encountered in pursuing neurosurgery training in the African context. METHODS: A cross-sectional electronic survey, developed in English and subsequently translated into French, was utilized. This was disseminated among medical students, medical interns, and junior doctors in all 5 African regions. RESULTS: A total of 491 responses were received from the following 5 African regions: West Africa (30.5%), East Africa (30.3%), Central Africa (18.1%), North Africa (11.8%), and Southern Africa (9.2%). Of the participants, 55.2% were men and 44.8% were women. Seventy-six percent of respondents were aware of the neurosurgery deficit in Africa. Barriers to pursuing neurosurgery included discouragement from others over lack of an "ideal personality" trait, culture/cultural beliefs as related to gender roles, lack of mentorship, inadequate neurosurgical training opportunities, poor health infrastructure for neurosurgical practice in Africa, poor patient outcomes, and challenges with work/life balance were some of the barriers highlighted to pursuing neurosurgical career on the continent. CONCLUSION: Even with its robust population growth, Africa is not producing enough neurosurgeons to meet the demands of the population due to several barriers. Delineating these challenges and barriers represents an important step in developing sustainable mechanisms for recruitment, training, mentorship, and support of burgeoning African neurosurgeons.

The landscape of neuro-oncology in East Africa: a review of published records.

INTRODUCTION: Primary central nervous system (CNS) tumors represent an important and growing cause of worldwide morbidity and mortality. There are global variations in the reported case burden of CNS tumors, with high-income countries reporting a higher incidence of cases than low- and middle-income countries. Variations are attributed to differences in access to care, diagnostic capacity, risk exposure, and under-reporting in LMICs. This study aims to review existing literature on the distribution of primary CNS tumors and neuro-oncologic care, and the contribution of scientists and institutions to neuro-oncologic research across 18 East African countries over the last 5 decades. METHOD: A search was conducted using OVID Medline and PubMed databases to identify relevant East African neuro-oncologic studies published over the last 50 years. RESULTS: The authors reviewed 36 neuro-oncology articles published across 8 of 18 East African countries. Kenya represented the highest number of published articles; ten countries queried yielded zero publications. A total of 2006 cases from all age groups were represented in published literature consisting of a wide spectrum of CNS tumors. One-third of reported cases were pediatric. Meningioma formed the largest proportion (43.3%) followed by glioma (33.7%). Sixty-seven percent of publications gave an overview of clinical care received by patients with most patients not receiving comprehensive neuro-oncologic care. CONCLUSION: The modest collection of neuro-oncology publications from East Africa shows that the case diversity of primary CNS tumors in East Africa is comparable to the rest of the world. There is, however, poorer access to neurosurgical care and adjuvant therapy. Multidisciplinary efforts from clinicians, researchers, and healthcare agencies are needed to quantify and address the requisite neuro-oncology needs in this region.

Disseminated craniospinal low-grade glioma in a patient with NF-1 without optic pathway pathology: illustrative case.

BACKGROUND: Neurofibromatosis type 1 (NF-1) is a neurocutaneous autosomal dominant disorder that predisposes patients to develop intracranial low-grade gliomas (LGGs). Most LGGs in patients with NF-1 involve the optic pathway but can arise anywhere throughout the central nervous system. NF-1-related disseminated pediatric LGG (dPLGG) in the absence of a dominant optic pathway glioma has not been described. OBSERVATIONS: The authors discussed a case of a 10-year-old boy who presented with consideration for biopsy with nonoptic pathway PLGG with craniospinal dPLGG in the setting of NF-1. The patient's primary lesion, located in the right medulla, was initially treated with surveillance before induction chemotherapy with carboplatin and vincristine was initiated. However, surveillance imaging demonstrated significant increase in size and enhancement, and subsequent craniospinal imaging demonstrated extensive nodular dissemination in the cervicothoracic spine. A biopsy and molecular testing were subsequently performed to further evaluate the tumor, and the patient was diagnosed with dPLGG with CDKN2A deletion. LESSONS: Thorough craniospinal magnetic resonance imaging evaluation and biopsy in nonoptic pathway-dominant brain lesions in NF-1 are warranted in patients with atypical clinical and radiological findings in whom standard chemotherapeutic therapy fails.

Racial and ethnic disparities in survival of children with brain and central nervous tumors in the United States.

BACKGROUND: Despite improvements in overall survival for pediatric cancers, treatment disparities remain for racial/ethnic minorities compared to non-Hispanic Whites; however, the impact of race on treatment outcomes for pediatric brain and central nervous system (CNS) tumors in the United States is not well known. METHODS: We included 8713 children aged 0-19 years with newly diagnosed primary brain and CNS tumors between 2000 and 2015 from the Census Tract-level SES and Rurality Database developed by Surveillance, Epidemiology, and End Results (SEER) Program. We used chi-square tests to assess differences in sociodemographic, cancer, and treatment characteristics by race/ethnicity and Kaplan-Meier curves and Cox proportional hazards models to examine differences in 10-year survival, adjusting for these characteristics. RESULTS: Among 8713 patients, 56.75% were non-Hispanic White, 9.59% non-Hispanic Black, 25.46% Hispanic, and 8.19% from "other" racial/ethnic groups. Median unadjusted survival for all pediatric brain tumors was 53 months, but varied significantly by race/ethnicity with a median survival of 62 months for non-Hispanic Whites, 41 months for non-Hispanic Blacks, and 40 months for Hispanic and other. Multivariable analyses demonstrated minority racial groups still had significantly higher hazard of death than non-Hispanic Whites; Hispanic (adjusted hazard ratio [aHR] 1.25 [1.18-1.31]); non-Hispanic Black (aHR 1.12 [1.04-1.21]); other (aHR 1.22 [1.12-1.32]). Results were consistent when stratified by tumor histology. CONCLUSION: We identified disparities in survival among racial/ethnic minorities with pediatric brain and CNS tumors, with Hispanic patients having the highest risk of mortality. Eliminating these disparities requires commitment toward promoting heath equity and personalized cancer treatment.

Barriers to malaria prevention among immigrant travelers in the United States who visit friends and relatives in sub-Saharan Africa: A cross-sectional, multi-setting survey of knowledge, attitudes, and practices.

BACKGROUND: Despite achievements in the reduction of malaria globally, imported malaria cases to the United States by returning international travelers continue to increase. Immigrants to the United States from sub-Saharan Africa (SSA) who then travel back to their homelands to visit friends and relatives (VFRs) experience a disproportionate burden of malaria illness. Various studies have explored barriers to malaria prevention among VFRs and non-VFRs-travelers to the same destinations with other purpose for travel-but few employed robust epidemiologic study designs or performed comparative analyses of these two groups. To better quantify the key barriers that VFRs face to implement effective malaria prevention measures, we conducted a comprehensive community-based, cross-sectional, survey to identify differences in malaria prevention knowledge, attitudes, and practices (KAP) among VFRs and others traveling to Africa and describe the differences between VFRs and other types of international travelers. METHODS AND FINDINGS: Three distinct populations of travelers with past or planned travel to malaria-endemic countries of SSA were surveyed: VFRs diagnosed with malaria as reported through a state health department; members of the general VFR population (community); and VFR and non-VFR travelers presenting to a travel health clinic, both before their pretravel consultation and again, after return from travel. A Community Advisory Board of African immigrants and prior qualitative research informed survey development and dissemination. Across the three groups, 489 travelers completed surveys: 351 VFRs and 138 non-VFRs. VFRs who reported taking antimalarials on their last trip rated their concern about malaria higher than those who did not. Having taken five or more trips to SSA was reported more commonly among VFRs diagnosed with malaria than community VFRs (44.0% versus 20.4%; p = 0.008). Among travel health clinic patients surveyed before and after travel, VFR travelers were less successful than non-VFRs in adhering to their planned use of antimalarials (82.2% versus 98.7%; p = 0.001) and employing mosquito bite avoidance techniques (e.g., using bed nets: 56.8% versus 81.8%; p = 0.009). VFRs who visited the travel health clinic were more likely than VFR respondents from the community to report taking an antimalarial (83.0% versus 61.9%; p = 0.009), or to report bite avoidance behaviors (e.g., staying indoors when mosquitoes were out: 80.9% versus 59.5%; p = 0.009). CONCLUSIONS: We observed heterogeneity in malaria prevention behaviors among VFRs and between VFR and non-VFR traveler populations. Although VFRs attending the travel health clinic appear to demonstrate better adherence to malaria prevention measures than VFR counterparts surveyed in the community, specialized pretravel care is not sufficient to ensure chemoprophylaxis use and bite avoidance behaviors among VFRs. Even when seeking specialized pretravel care, VFRs experience greater barriers to the use of malaria prevention than non-VFRs. Addressing access to health care and upstream barrier reduction strategies that make intended prevention more achievable, affordable, easier, and resonant among VFRs may improve malaria prevention intervention effectiveness.