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We report the first case of human T-lymphotropic type 1-associated myelopathy (HAM) linked with primary sclerosing cholangitis (PSC). The patient, a 44-year-old woman, also suffered from ulcerative colitis and polyarthritis. Two years and nine months after the clinical onset of PSC, the coexistence of HAM was shown by neurological manifestations characterized by slowly progressive symmetrical myelopathy, predominantly involving pyramidal tracts, and positive antibodies to human T-lymphotropic virus type 1 in serum as well as in cerebrospinal fluid.
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