Study Design: Single subject (male, 34 years-of-age) case report. Objectives: To describe a unique case of plasma cell granuloma (PCG) with simultaneous brain and spinal cord involvement. Setting: Private hospital, capital city, Turkey. Methods: The patient presented with headache and paraplega. T2 weighted MR images showed multiple hyperintense cerebral, cerebellar, brainstem and spinal cord lesions that enhanced homogeneously with contrast medium. The patient was operated on, and the two intramedullary spinal cord lesions at T11 and T12 were totally excised. Results: After 3 months of an intense physiotherapy programme the patient was able to walk with help. Conclusion: We report for the ®rst time, the occurrence of multiple, simultaneous brain and intramedullary spinal cord plasma cell granulomas.