• Virginie Hean, Wafa Bouleftour, Carole Ramirez, Fabien Forest, Claire Boutet, and Romain Rivoirard.
• Service de Neurologie, CHU de Saint-Etienne, Hôpital Nord, Avenue Albert Raimond, Saint Etienne CEDEX 2.
• Medicine (Baltimore). 2021 May 14; 100 (19): e25862e25862.

RationaleMeningeal melanocytoma is a rare benign melanocytic tumor of the central nervous system. We report for the first time a case of meningeal melanocytoma treated with immunotherapy.Patient ConcernsA 70-year-old man with no medical history was admitted to the Emergency Room. He suffered from a motor and sensory deficit in his left lower limb and a bilateral upper arm neuralgia.DiagnosesA contrast-enhanced magnetic resonance imaging (MRI) was performed. It showed a C7-T1 bleeding intramedullary tumor. Laminectomy was decided and performed. The results of the pathologic examination showed a melanocytic tumor harboring GNAQ mutation. Meningeal melanocytoma was the final diagnosis.InterventionsThe patient was treated with 10 radiotherapy sessions and 6 cycles of nivolumab. A year later, the patient experienced neuralgia again with severe pain and an increasing sensory motor deficit. He underwent a second surgery that was incomplete. As the tumor kept growing, he received temozolomide. But the 6th cycle had to be interrupted due to bedsore infection in the hip area.OutcomesDisease progression finally led to the patient's death 3 years after diagnosis.LessonsThis case report is the first about a patient with meningeal melanocytoma treated with immunotherapy. Treatment based on biomolecular mutations will probably change spinal melanocytoma therapeutic approach in the next few years.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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