Rheumatoid arthritis (RA) is a chronic inflammatory disease characterized mainly by arthritis, with the possible occurrence of extra-articular manifestations. We report the case of a patient who developed leptomeningitis as the first sign of RA, one year before the diagnosis of RA. Methylprednisolone 1000 mg was given intravenously. Because of the onset of seizures and cognitive impairment, rituximab was started. After three cycles of rituximab (1000 mg on day 0 and 1000 mg on day 15, every 6 months), neurological clinical examination, MRI and electroencephalogram findings were significantly improved.

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