SynthesisChild's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery2026
Impact of chemotherapy and immunotherapy on survival in pediatric primary diffuse leptomeningeal melanomatosis: a systematic review and pooled survival analysis.
Synthesis in Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
purposePediatric primary diffuse leptomeningeal melanomatosis (PDLM) is a rare, aggressive malignancy with no established standard of care. We performed an integrated survival analysis to characterize disease course and identify treatment factors associated with overall survival (OS).
methodsA systematic literature search of PubMed, Embase, and Scopus (2000-2025) was conducted in accordance with PRISMA guidelines. Eligible studies included patients < 18 years with histopathologically confirmed PDLM per 2021 World Health Organization and Expert Care for Rare Adult Solid Cancers criteria and reported OS and treatment data. Survival was assessed using Kaplan-Meier analyses, with univariable and multivariable Cox proportional hazards regression.
resultsThirty-three pediatric PDLM cases were identified (57.6% male; median age 10 [IQR 3-14] years). Diagnosis was established via tissue sampling in 72.7%, cerebrospinal fluid cytology in 21.2%, and both in 6.1%. Among 13 patients with molecular testing, 69.2% harbored NRAS mutations. Twenty-four patients (72.7%) received treatment: chemotherapy (70.7%), radiation therapy (41.7%), and immunotherapy (62.5%). Median OS was 5.0 [3.5-7.0] months. On Kaplan-Meier analysis, immunotherapy was associated with improved OS (p < 0.001), including when combined with radiation (p = 0.038), chemotherapy (p = 0.006), or both (p = 0.048). Univariable Cox regression showed immunotherapy to be significantly associated with reduced hazard of death (HR 0.23 [0.10-0.51], p < 0.001). On multivariable analysis, immunotherapy (HR 0.22 [0.10-0.49], p < 0.001) and chemotherapy (HR 0.44 [0.21-0.94], p = 0.035) were independently associated with improved OS.
conclusionPediatric PDLM carries a uniformly poor prognosis. Immunotherapy and chemotherapy were the only treatments independently associated with improved survival, highlighting their central role in management of this rare disease.
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