Abstract Background Systemic immune dysregulation is well-characterized in adult high-grade gliomas but remains underexplored in pediatric population. Among these, H3K27M-mutant diffuse midline gliomas are biologically and clinically distinct, with limited therapeutic options and poor prognosis. While prior studies in medulloblastoma suggest tumor-induced lymphopenia at diagnosis, the immune profile of H3K27M gliomas has not been systematically evaluated. Methods We performed a multicenter retrospective analysis of pretreatment complete blood counts (CBCs) in pediatric patients (≤18 years) with H3K27M-mutant gliomas (n = 37), comparing them to a control cohort with pilocytic astrocytoma (n = 61). Absolute neutrophil count (ANC), monocyte count (AMC), lymphocyte count (ALC), neutrophil-to-lymphocyte count ratio (NLCR), and monocyte-to-lymphocyte ratio (MLR) were evaluated. Patients receiving corticosteroids prior to CBC collection were excluded. Results No significant differences were observed in ANC, ALC, or NLCR between groups. However, the H3K27M group demonstrated significantly elevated AMC (0.70 vs. 0.50 K/μL; p = 0.012) and MLR (0.20 vs. 0.19; p = 0.021) compared to controls. Monocytosis, defined by AMC above age-adjusted means, was present in 78.4% of H3K27M patients versus 46.7% of controls (OR = 4.14, p = 0.003). These findings suggest a distinct pre-treatment immune signature in H3K27M gliomas. Conclusions This is the first study to identify pre-treatment monocytosis and elevated MLR in pediatric H3K27M-mutant gliomas, highlighting a novel systemic immune phenotype with potential implications for biomarker discovery and immunotherapeutic targeting. These results underscore the need for further mechanistic studies and prospective validation in larger, mutation-stratified cohorts.
BACKGROUND AND OBJECTIVES:Multiple-choice questions are the primary assessment format for neurosurgical board certification. Creating high-quality examination questions requires significant expert time and resources. The goal of this study was to develop an automated system to generate board-style neurosurgical multiple-choice questions using state-of-the-art vision-language models and compare their quality with authentic self-assessment questions. METHODS:We developed an automated pipeline using OpenAI generative pre-trained transformer (GPT)-4o and Anthropic Claude Sonnet-3.5 to generate neurosurgical board-style questions from Neurosurgery Publications articles. We generated 89 587 synthetic questions: 45 689 with GPT-4o and 43 898 with Claude. Each question was associated with a single image extracted from the articles' figures. We evaluated the quality of synthetic questions through 5 surveys comparing 20 synthetic questions (10 from each model) with 10 authentic questions from the Self-Assessment for Neurological Surgeons (SANS) question bank. Each survey was completed by a neurosurgery resident and an attending who guessed the source [human vs artificial intelligence (AI)-generated] and rated suitability for board examination use. We also evaluated the question-answering performance of the generalist GPT-4o and the specialized CNS-Obsidian. RESULTS:SANS questions were more often perceived as human-made than GPT-generated (residents, P = .0002; attendings, P = .1091) and Claude-generated (residents, P = .0002; attendings, P = .0272) questions. Notably, 54% of AI-generated questions misled at least one evaluator, and 23% misled both. In quality assessments, SANS questions outperformed GPT-generated (residents, P < 10-5; attendings, P = .0001) and Claude-generated (residents and attendings, P < 10-5) questions. Particularly, 25% of AI-generated questions were rated as suitable for board examinations vs 72% of human-generated questions when measured by evaluator consensus (P < 10-7). CONCLUSION:Although quality gaps exist between AI-generated and human-created neurosurgical board examination questions, our approach demonstrates the potential of vision-language models to augment assessment development in specialized medical fields, reducing the burden on examination boards and credentialing organizations.
PURPOSE:Pediatric low-grade gliomas (pLGGs) typically have excellent long-term outcomes; intratumoral hemorrhage is a rare, potentially dangerous complication. Hemorrhage risk in the context of molecular alterations and targeted therapies remains poorly characterized. We analyzed the incidence, timing, and independent risk factors for hemorrhage in a large contemporary pLGG cohort. METHODS:We conducted a retrospective cohort study of 236 children with pLGG treated at a single center (2011-2025). Clinical, radiographic, and molecular variables were abstracted. The primary endpoint was spontaneous tumoral hemorrhage. Time-to-event analyses utilized Kaplan-Meier methods and Cox proportional hazards modeling; penalized regression mitigated overfitting given the event rarity. RESULTS:Twelve patients (5.1%) experienced hemorrhage over 2,234 person-years (incidence: 0.54/100 person-years). Hemorrhage typically occurred years after initial tumor diagnosis (median 6.4 years). The presence of a KIAA1549::BRAF fusion in the tumor had the strongest association with hemorrhage, persisting across multivariable models (approximately sixfold increased risk), although estimates were limited by low event number. MAPK inhibitor exposure (specifically binimetinib and tovorafenib) was associated with hemorrhage in univariate analysis but partially confounded by fusion status. CSF diversion independently increased risk at brainstem, optic pathway, and hypothalamic locations. No hemorrhages occurred among patients with underlying genetic syndromes, including neurofibromatosis type 1 and tuberous sclerosis. CONCLUSION:Hemorrhage in pLGG is an infrequent late complication associated with tumor biology. KIAA1549::BRAF fusion may identify a higher-risk subgroup, with MAPK inhibitor exposure and CSF diversion further modifying risk. These findings support biology-informed surveillance and personalized management strategies for at-risk children.
BACKGROUND AND OBJECTIVES:Cerebrospinal fluid shunting is a common treatment for hydrocephalus. Several techniques for ventricular catheter placement exist, yet comparative safety data remain limited. This study evaluated whether stereotactic navigation-assisted ventricular catheter placement is associated with reduced 30-day postoperative intracranial hemorrhage (ICH) rates. METHODS:This was a retrospective analysis of adults who underwent frontal or occipital shunt placement for hydrocephalus between 2014 and 2023. The primary exposure variable was the use of stereotactic navigation for ventricular catheter placement. The primary outcomes were hospital length of stay (LOS) and 30-day postoperative ICH. RESULTS:A total of 541 shunt placements were included (median age 72.5 years; 51.6% male). Normal pressure hydrocephalus represented the most common underlying diagnosis (60.3%). Stereotactic navigation was used in 221 cases (40.9%) and was more frequently employed in younger patients (66.0 vs 73.9 years; P < .001), more recent years (median 2021 vs 2019; P < .001), and cases involving non-normal pressure hydrocephalus diagnoses (P < .001). Overall, 12 patients (2.2%) developed ICH within 30 days of surgery. The 30-day ICH rate was significantly lower with stereotactic navigation (0.5% vs 3.4%; odds ratio 0.13, 95% CI 0.02-1.00; P = .033), corresponding to an absolute risk reduction of 2.99% and a number needed to treat of 34. Kaplan-Meier analysis demonstrated superior hemorrhage-free survival with navigation-assisted placement (log-rank P = .021), with most events occurring within the first 14 days postoperatively. Propensity-matched analysis demonstrated a similar effect size but fell short of statistical significance. Navigation use was not independently associated with LOS or any other complications. CONCLUSION:Stereotactic navigation-assisted ventricular catheter placement was associated with lower rates of 30-day postoperative ICH compared with non-navigation-assisted placement, with a number needed to treat of 34. Given the absence of adverse effects on LOS or overall complication rates, these findings support routine consideration of neuronavigation during shunt placement when available.
BACKGROUND AND OBJECTIVES:General purpose vision-language models (VLMs) demonstrate impressive capabilities, but their opaque training on uncurated internet data poses critical limitations for high-stakes decision making, such as in neurosurgery. We present CNS-Obsidian, a neurosurgical VLM trained on peer-reviewed neurosurgical literature, and demonstrate its clinical utility compared with GPT-4o in a real-world setting. METHODS:We compiled 23 984 articles from Neurosurgery Publications journals, yielding 78 853 figures and captions. Using GPT-4o and Claude Sonnet-3.5, we converted these image-text pairs into 263 064 training samples across 3 formats: instruction fine-tuning, multiple-choice questions, and differential diagnosis. We trained CNS-Obsidian, a fine-tune of the 34-billion parameter Large Language and Visual Assistant-Next model. In a blinded, randomized deployment trial at NYU Langone Health (August 30-November 30, 2024), neurosurgeons were assigned to use either CNS-Obsidian or a Health Insurance Portability and Accountability Act-compliant GPT-4o end point as a diagnostic copilot after patient consultations. Primary outcomes were diagnostic helpfulness and accuracy, assessed through user ratings and presence of the correct diagnosis within the VLM-provided differential, respectively. RESULTS:CNS-Obsidian matched GPT-4o on synthetic questions (76.13% vs 77.54%, P = .235), but only achieved 46.81% accuracy on human-generated questions vs GPT-4o's 65.70% (P < 10-15). In the randomized trial, 70 consultations were evaluated (32 CNS-Obsidian, 38 GPT-4o) from 959 total consults (7.3% utilization). CNS-Obsidian received positive ratings in 40.62% of cases vs 57.89% for GPT-4o (P = .230). Both models included correct diagnosis in approximately 60% of cases (59.38% vs 65.79%, P = .626). CONCLUSION:Domain-specific VLMs trained on curated scientific literature can approach frontier model performance in specialized medical domains despite being orders of magnitude smaller and less expensive to train. This establishes a transparent framework for scientific communities to build specialized artificial intelligence models. However, low clinical utilization suggests chatbot interfaces may not align with specialist workflows, indicating need for alternative artificial intelligence integration strategies.
Abstract Background Diffuse intrinsic pontine glioma (DIPG) can cause secondary cerebellar tonsillar descent, resulting in a coexisting Chiari malformation (CM). Because both conditions involve the posterior fossa, their symptom profiles overlap, making it difficult to separate Chiari-related morbidity from tumor effects. The clinical relevance of this dual pathology remains uncertain. This study aimed to define the incidence, clinical impact, management, and evolution of CM in children with DIPG. Methods We performed a retrospective cohort study of pediatric patients with DIPG (2016–2024) to assess the incidence, presentation, and management of associated CM. Results Thirty-six patients with DIPG (median age 4.9 years, range 2.1–19.1) met inclusion criteria. CM was present in 7 patients (19.4%). Radiographic CM appeared a mean of 6 months (range 0–29) after diagnosis, with four cases (57.1%) present at initial imaging. Median tonsillar position was 7 mm (IQR 6.0). Five patients (71.4%) were symptomatic with classic CM symptoms. Six (85.7%) developed hydrocephalus, and one had a syrinx. CM resolved spontaneously in 5 patients (71.4%), remained stable in one, and progressed in one. CM incidence was not associated with age, sex, hydrocephalus, or CSF diversion (p > 0.05). Although a smaller dens angle correlated with CM (p = 0.039), the 3-degree median difference was of limited clinical significance. Conclusions Nearly one in five children with DIPG exhibit tonsillar descent meeting criteria for CM. No patient required Chiari-directed surgical decompression; CM usually resolved spontaneously. These findings suggest that DIPG-associated CM rarely merits independent intervention and may not increase hydrocephalus risk.
BACKGROUND AND OBJECTIVES:Normal pressure hydrocephalus (NPH) affects approximately 0.2% to 2.9% of adults older than 65 years and is characterized by progressive gait impairment, cognitive decline, and urinary incontinence. Cerebrospinal fluid shunting is the mainstay of treatment but carries a risk of overdrainage, leading to subdural collections. Evidence guiding the management of these collections in patients with shunted NPH remains limited. We aimed to characterize treatment strategies and outcomes for subdural collections in patients with shunted NPH and to propose a practical management framework for nonemergent presentations. METHODS:We performed a retrospective cohort study of patients with shunted NPH who developed subdural collections requiring treatment between January 2014 and September 2025. Interventions included valve pressure adjustment, middle meningeal artery (MMA) embolization, and surgical evacuation. Clinical, radiographic, and treatment data were extracted from the electronic health record. Time-to-resolution and recurrence were assessed using Kaplan-Meier analysis. Receiver operating characteristic analysis determined the predictive value of baseline collection volume for failure of valve adjustment. RESULTS:Sixty patients with 96 total collections underwent 91 valve pressure increases, 8 craniotomies for 8 collections, 23 MMA embolization procedures for 32 collections, and 15 subdural evacuating port system procedures for 16 collections. The median time to resolution for collections that underwent valve adjustment only, MMA embolization without evacuation, evacuation without MMA embolization, and both MMA embolization and evacuation was 1.91, 3.99, 1.77, and 5.17 months, respectively. Using pretreatment volume to predict valve adjustment failure demonstrated an area under the curve of 0.75 with an optimal cutoff of 40.05 cm3, corresponding to a sensitivity of 75.0% and a specificity of 71.4%. CONCLUSION:Baseline hematoma volume may help identify which shunted NPH patients with subdural hematomas are at higher risk of failing valve adjustment alone. We propose a volume-informed management framework in which larger hematomas (approximately >40 cm3) prompt earlier consideration of treatment escalation.
OBJECTIVE:Hydrocephalus secondary to CNS tumors is common and carries high shunt failure rates, yet predictors of shunt survival remain poorly defined. The aim of this study was to identify patient-, tumor-, and treatment-related predictors of shunt outcomes in neoplastic hydrocephalus. METHODS:This retrospective single-institution analysis included consecutive patients who underwent ventriculoperitoneal or ventriculoatrial shunt placement for neoplastic hydrocephalus between February 2013 and February 2024. Patients were identified using validated natural language processing and manual review. Demographic, oncological, and surgical variables, including tumor location, neurological symptoms, and resection history, were analyzed. The primary outcome was shunt revision for any cause at 1, 3, and 5 years. Fisher's exact test or the chi-square test was used for categorical comparisons. Kaplan-Meier and log-rank analyses were used to assess shunt survival, with subgroup analysis for age and etiology. RESULTS:Overall, 239 patients (140 female, median age 53 years) were included, with 186 adults and 53 pediatric patients. Intraventricular tumor location showed the strongest association with shunt failure across all time points (5-year OR 4.39, p < 0.001). Preoperative neurological symptoms (seizures and visual/auditory deficits) were associated with higher odds of single and multiple shunt revisions (OR range 1.92-2.15, p ≤ 0.03). WHO grade 2 tumors trended toward greater late (5-year) failure risk (OR 2.8, p = 0.06). Obstruction accounted for 35.6% of shunt failures, followed by infection. Adjuvant chemotherapy and radiation therapy were not associated with shunt failure after adjustment for survival differences. CONCLUSIONS:In this largest-to-date cohort of patients who underwent shunt placement for neoplastic hydrocephalus, the factors associated with shunt failure were intraventricular tumor location, multiple resections, and preoperative neurological symptoms. Adjuvant therapies had minimal impact. These findings support individualized risk stratification and targeted follow-up and provide a foundation for targeted prospective studies.
BACKGROUND AND OBJECTIVES:Normal pressure hydrocephalus (NPH) is characterized by the classic triad of cognitive decline, gait instability, and urinary incontinence in the setting of ventriculomegaly with normal intracranial pressure. Cerebrospinal fluid diversion is the current standard treatment, yet it carries a risk of overdrainage, resulting in subdural hematoma or hygroma. Different valves have been developed to mitigate this risk, yet consensus remains unclear regarding optimal valve for NPH. METHODS:We performed a retrospective cohort study on all patients with NPH who underwent cerebrospinal fluid shunting or revision between January 2014 and September 2025 at our institution. Demographic, clinical, and radiological data were collected from the electronic health record. Kaplan-Meier survival analysis, univariate logistic regression, and multivariate modeling were used to identify predictors of subdural collections and the need for surgical treatment. RESULTS:Since our change in practice from the Integra NPH Low Flow Valve (Low Flow OSV) to other valves in 2022, we observed a rise in symptomatic subdural collections. Programmable valves were associated with a markedly increased 1-year risk of both subdural collection formation and need for surgical intervention compared with the Low Flow OSV. Overall, Certas and Strata valves demonstrated higher rates of subdural collections requiring surgery than the Low Flow OSV (14.6% vs 2.1%, P < .001; 10.5% vs 2.1%, P = .005, respectively). On multivariate analysis, both the Strata and Certas valves were independently associated with increased odds of developing any subdural collection and necessitating surgery. Vascular disease and dual antiplatelet therapy also increased risk. CONCLUSION:In this large single-center cohort study, programmable valves, specifically the Certas and Strata, were associated with an increased rate and severity of subdural collections compared with the Low Flow OSV. The use of low-flow designs may mitigate complications for the NPH population, and the use of lower programmable valve settings should be carefully considered.
Abstract Introduction Pediatric low-grade gliomas (pLGGs) are the most common childhood brain tumors and are generally associated with excellent long-term survival. However, intratumoral hemorrhage represents a rare but potentially devastating complication. Existing data on hemorrhage risk in pLGG are limited to small, heterogeneous series, and the impact of molecular alterations and modern targeted therapies remains poorly defined. We sought to determine the incidence, timing, and independent risk factors for hemorrhage in a large contemporary pLGG cohort. Methods We conducted a retrospective cohort study of 236 patients with pLGG treated at a single academic center between 2011 and 2025. Clinical, radiographic, molecular, and treatment-related variables were systematically abstracted. The primary endpoint was spontaneous tumoral hemorrhage. Time-to-event analyses were performed in Python using Kaplan–Meier methods and Cox proportional hazards modeling. Given the rarity of hemorrhage events, parsimonious multivariable models and penalized Cox regression were used to mitigate overfitting. Results Twelve patients (5.1%) experienced hemorrhage over 2,234 person-years of follow-up, yielding an incidence rate of 0.54 per 100 person-years. Hemorrhage typically occurred years after initial tumor diagnosis (median 6.4 years). The presence of a BRAF–KIAA1549 fusion in the tumor was the strongest independent predictor of hemorrhage across all multivariable models, conferring a sixfold increased risk. MAPK inhibitor exposure, particularly binimetinib and tovorafenib, was associated with hemorrhage in univariate analysis, though partially confounded by fusion status. Previous CSF diversion procedures independently increased hemorrhage risk in tumors located in the brainstem, optic pathway, or hypothalamus compared to other locations. No hemorrhages occurred among patients with underlying genetic syndromes, including neurofibromatosis type 1 and tuberous sclerosis. Conclusion Hemorrhage in pLGG is an uncommon but late complication driven primarily by tumor biology. BRAF–KIAA1549 fusion identifies a high-risk subgroup in whom treatment-related and procedural factors further modify risk, enabling biology-informed surveillance and management strategies.
Traumatic brain injury (TBI) is the leading cause of pediatric disability. Most pediatric TBIs are mild but can result in long-term cognitive and functional impairments. Outpatient follow-up is essential to detect post-concussive symptoms and aid recovery. All patients 3–18 years of age with positive TBI findings on CT or MRI from 2018–2024 were retrospectively reviewed. Follow-up was defined as an appointment with neurology, neuropsychology, neurosurgery, or physical medicine and rehabilitation within three months of discharge. Analyses were performed with appropriate Chi-squared, Fisher’s exact, Mann–Whitney U, or t-tests. Fifty-seven patients (41 male, mean age 11.4 years) were identified, with mild TBIs (GCS 13–15) comprising 41/57 (71.9
INTRODUCTION:Hemispherotomy is an effective treatment for children with drug-resistant epilepsy (DRE). While hemispherotomy techniques and indications have evolved, access remains predominantly constrained to high-resource settings. METHODS:We performed a retrospective analysis of children who underwent hemispherotomy from 2011 to 2023 by a hybrid team, including local Panamanian and US neurologists, neurosurgeons, and EEG technicians and analyzed surgical, epilepsy, and quality of life (QoL) parameters. Follow-up data were collected according to the International Consortium for Health Outcomes Measurement (ICHOM) guidelines for children with epilepsy. RESULTS:Twenty-three children underwent hemispherotomy. The median age at surgery was 10 years (range 2-20). The median follow-up time was 6 years (range 1-13). The etiology of DRE included malformations of cortical development in 14 children (60.8%), including 8 (34.8%) with schizencephaly, and secondary causes in 9 children (39.1%). Seizure frequency improved for all 23 children (100%): Engel I was achieved in 15 children (65.2%), Engel II (26.1%) in six children, and Engel III (8.7%) in two children. Patients with seizure freedom had significantly fewer preoperative seizures per day than patients with seizure recurrence. Complications occurred in six children (26.1%): 2 wound infections, 2 meningitis, 1 femoral vein thrombosis, and 1 wound hematoma with return to OR. There were no perioperative mortality and no postoperative hydrocephalus or CSF diversion. QoL-related outcomes were available for 16 children: 16/16 (100%) reported that the surgery was a worthwhile and repeatable choice, 14 (87.5%) reported improved cognitive function, the median QOLCE-16 score was 62.5 ± 21. CONCLUSION:Hemispherotomy for DRE in selected children is a safe and effective surgery in a public children's hospital in a low-resource setting. At last follow-up, the majority of children were seizure-free, and all children had decreased seizure frequency. Families reported improved cognitive function, improved QoL and high satisfaction with their decision to pursue this surgery.
To identify clinical variables associated with ventricular shunt infection and shunt failure in pediatric hydrocephalus. Patients ≤ 18 years treated with ventricular shunts between 2013 and 2024 were identified from one institution’s electronic medical record. Children with a confirmed diagnosis of hydrocephalus and ≥ 6 months of postoperative follow-up were included. Primary and revision shunt surgeries were included. Records were manually reviewed for clinical variables. Statistical analyses were performed using R (version 4.2.3). The dataset included 474 surgeries, 146 primary and 328 revisions, undergone by 226 patients. Infection necessitating removal of a previously placed shunt occurred following 3.59
In children undergoing craniotomy, the impact of postoperative pain on recovery is receiving growing recognition. While opioids are often the primary treatment, their administration requires a delicate balance between achieving sufficient analgesia and mitigating side effects like sedation, nausea, vomiting, and respiratory depression. We review the emerging adjunct treatment modality regional scalp block (RSB) infiltration for post-craniotomy pain. Postoperative pain after pediatric craniotomy can be challenging to manage and may contribute to unnecessary suffering as well as the development of long-term neurocognitive and psychological sequelae. Pain during the PICU stay is also a major risk factor for post-PICU syndrome, which involves persistent impairments in children’s physical, cognitive, or mental health persisting beyond acute hospitalization. Despite increasing awareness and treatment strategies for post-craniotomy pain in adults, significant gaps remain in understanding its assessment and management in children. This review examines the current literature surrounding post-craniotomy pain management in children with a special emphasis on RSB, a treatment option increasingly used in adults and children. RSB has been shown in randomized trials to reduce postoperative pain and opioid use. However, while pediatric perioperative trials support its safety and feasibility, robust clinical evidence supporting RSB’s efficacy for post-craniotomy pain in children remains limited, hindering wider translation into clinical standard. RSB infiltration is an emerging and promising technique for pediatric post-craniotomy pain management. Early evidence suggests it is both safe and effective, with potential to enhance postoperative recovery and to be integrated into clinical practice. Further research is critical to validate initial findings and better define the benefits across diverse pediatric populations.
BACKGROUND:Sturge-Weber syndrome (SWS) is a congenital neurocutaneous disorder characterized by angiomas of the face, choroid, and leptomeninges. Seizures in these children often present within the first 2 years of life. SWS is typically unilateral, but bilateral SWS occurs in approximately 15% of cases. Bilateral SWS is associated with earlier seizure onset and poorer cognitive, developmental, and functional outcomes. More than half of children with SWS develop drug-resistant epilepsy requiring surgical intervention. Hemispherotomy has been established as a successful treatment for unilateral SWS, but resective surgery has traditionally not been considered a treatment option for patients with bilateral disease. OBSERVATIONS:In this report, the authors present the cases of 4 children (7 months-2 years of age) with bilateral SWS and drug-resistant epilepsy with a unilateral electroencephalography predominance. After a multidisciplinary conference in each case, all children were successfully treated with unilateral hemispherotomy. These patients achieved prolonged periods of seizure freedom postoperatively, a better quality of life, and demonstrated improved developmental progress at long-term follow-up. LESSONS:This case series suggests that functional hemispherotomy may be a safe and effective therapeutic option for improving seizure burden in cases of bilateral drug-resistant SWS with asymmetric seizure burden. https://thejns.org/doi/10.3171/CASE25125.
BACKGROUND:Tuberous sclerosis complex (TSC) is a neurocutaneous disorder characterized by the multisystem development of benign tumors. Patients with TSC are also at an increased risk of developing intracranial aneurysms early in life. While aneurysms have historically been treated with open surgical clipping, endovascular approaches are increasingly being used in both pediatric and adult populations. OBSERVATIONS:In this case series, the authors report the endovascular treatment of three young patients with TSC and fusiform intracranial aneurysms using the Pipeline embolization device (PED) for flow diversion of the affected artery. In all cases, complete aneurysm occlusion was observed, with good parent artery wall reconstruction and no parent artery stenosis or occlusion. All flow diverter stents were deployed successfully, and there were no device-related complications. All patients were maintained on dual antiplatelet therapy. LESSONS:The PED can serve as a safe and effective endovascular alternative to open surgery for the treatment of intracranial aneurysms in young patients with TSC. This could avoid an additional craniotomy in a population already likely to require surgical intervention for TSC-related tumors or epilepsy. Further studies investigating the use of the PED in the pediatric population must be undertaken to validate its long-term efficacy. https://thejns.org/doi/10.3171/CASE24452.