Journal of neurosurgery. Pediatrics · 2026
Spinal intradural epidermoid cysts in pediatric patients: illustrative cases and systematic review.
СтатьяСистематический обзорКлинический случайНазвание и аннотация: Перевод в очереди
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Ibrahim Sufyan, Balasubramanian Kishore, Gulsuna Beste, Jea Andrew, Sader Nicholas
Аннотация
OBJECTIVE: Spinal intradural epidermoid cysts (sIECs) are rare, benign neoplasms comprising < 3% of intraspinal tumors in children. Their nonspecific clinical presentation and rarity often lead to diagnostic delays, particularly in the pediatric population. Authors of this study present 2 pediatric sIEC cases and conduct a systematic review of the literature to characterize the clinical features, management strategies, and outcomes of pediatric sIEC.
METHODS: A systematic review was conducted according to PRISMA guidelines, searching the PubMed, Embase, Web of Science, and Cochrane databases from inception to December 2024 for English-language case reports and case series describing pediatric sIEC with clinical, radiological, and follow-up data. Two illustrative pediatric cases managed at the authors' institution are also described.
RESULTS: Thirty-eight studies comprising 48 pediatric patients were included in the analysis, as were 2 cases from the authors' institution, yielding a pooled cohort of 50 pediatric patients. The median age at presentation was 7.5 years (IQR 9.75 years), with males representing 49% of the patients. Muscle weakness (78%), sensory deficits (38%), bowel or bladder dysfunction (38%), gait disturbance (34%), and back pain (30%) were common symptoms. Spinal dysraphism (48%) and infection (20%) were frequently associated with sIECs. Tumors most often involved the thoracic (30%) and lumbar (30%) spine, with 72% of cases spanning multiple levels. Gross-total resection (GTR) was achieved in 71% (34/48) of cases, and GTR significantly reduced recurrence (3% vs 29% for non-GTR, p = 0.03, OR 0.09, 95% CI 0.01-0.86). At the last follow-up (median 8 months), 64% (28/44) of patients were symptom free and 30% (13/44) experienced symptom improvement. The tumor recurred in 12% (6/50) of cases, and overall survival was 98% (49/50 patients). GTR with complete symptom resolution was achieved in both illustrative cases.
CONCLUSIONS: In this first systematic review addressing sIEC in children, patients typically presented with progressive neurological deficits and the cyst was frequently associated with congenital spinal anomalies. Diffusion-weighted imaging was essential for diagnosis and surgical planning. GTR was associated with significantly lower recurrence rates and favorable neurological outcomes.
METHODS: A systematic review was conducted according to PRISMA guidelines, searching the PubMed, Embase, Web of Science, and Cochrane databases from inception to December 2024 for English-language case reports and case series describing pediatric sIEC with clinical, radiological, and follow-up data. Two illustrative pediatric cases managed at the authors' institution are also described.
RESULTS: Thirty-eight studies comprising 48 pediatric patients were included in the analysis, as were 2 cases from the authors' institution, yielding a pooled cohort of 50 pediatric patients. The median age at presentation was 7.5 years (IQR 9.75 years), with males representing 49% of the patients. Muscle weakness (78%), sensory deficits (38%), bowel or bladder dysfunction (38%), gait disturbance (34%), and back pain (30%) were common symptoms. Spinal dysraphism (48%) and infection (20%) were frequently associated with sIECs. Tumors most often involved the thoracic (30%) and lumbar (30%) spine, with 72% of cases spanning multiple levels. Gross-total resection (GTR) was achieved in 71% (34/48) of cases, and GTR significantly reduced recurrence (3% vs 29% for non-GTR, p = 0.03, OR 0.09, 95% CI 0.01-0.86). At the last follow-up (median 8 months), 64% (28/44) of patients were symptom free and 30% (13/44) experienced symptom improvement. The tumor recurred in 12% (6/50) of cases, and overall survival was 98% (49/50 patients). GTR with complete symptom resolution was achieved in both illustrative cases.
CONCLUSIONS: In this first systematic review addressing sIEC in children, patients typically presented with progressive neurological deficits and the cyst was frequently associated with congenital spinal anomalies. Diffusion-weighted imaging was essential for diagnosis and surgical planning. GTR was associated with significantly lower recurrence rates and favorable neurological outcomes.