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We describe the clinical, radiologic, and postmortem findings of a 42-year-old man with intravascular lymphomatosis. The patient presented with a conus medullaris syndrome followed by progressive, disseminated spinal and cerebral symptoms. Disseminated encephalomyelitis was suspected due to the clinical, radiologic, and cerebrospinal fluid findings and the results of a stereotactic brain biopsy, all of which were compatible with inflammatory CNS disease. Treatment with methylprednisolone and cyclophosphamide led to a temporary remission of symptoms. The patient died 13 months after onset of symptoms. The diagnosis of disseminated intravascular lymphomatosis was established after death.