• Ana Misir Krpan, Zoran Rakusic, and Davorin Herceg.
• University Hospital Centre Zagreb, Kispaticeva 12.
• Medicine (Baltimore). 2020 Dec 11; 99 (50): e22928.

RationalePrimary leptomeningeal melanoma is an extremely rare disease of the central nervous system. There are no standard treatment protocols with a poor prognosis in very few reported cases. Immunotherapy in primary brain melanoma has not been successfully applied so far.Patient ConcernsWe describe a female patient 72-year-old diagnosed in the Neurosurgery Department which presented with generalized seizures.DiagnosesHistological examination confirmed atypical melanocytes immunohistochemically positive for melan A, HMB45 and S-100 protein in the meninges, BRAF V600E negative. Dermatological, ophthalmological examinations, and 18-FDG PET/CT were negative.InterventionsThe patient was successfully treated with pembrolizumab 2 mg/kg every 3 weeks for 2 years.OutcomesThe disease was stable for 2 years and the patient had no significant toxicity.LessonsOur report describes durable intracranial tumor response suggesting the efficacy of PD-1 inhibitor pembrolizumab for central nervous system primary leptomeningeal melanoma.

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