We report a 46-year-old-man who developed recurrent myelitis associated with hepatitis C viral (HCV) infection. Spinal cord biopsy showed acute demyelination without evidence of vasculitis. Antibodies to HCV were present in the CSF; HCV RNA was not detected in the CSF. Neither HCV antigens nor RNA were detected in the spinal cord biopsy, whereas they were found in the liver biopsy. Evaluation for other infectious or autoimmune causes was unrevealing. These observations suggest that recurrent myelitis in this patient is etiologically related to HCV infection, possibly via an immune-mediated mechanism. This is the first report of pathologically proven myelitis associated with HCV infection and we suggest that HCV be considered in the differential diagnosis of the transverse myelitis syndrome.