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The cases of seventy-two patients who had blastomycosis and were seen between 1926 and 1988 were retrospectively reviewed for involvement of the musculoskeletal system. Most patients were from rural rather than urban areas (almost a 2:1 ratio), and an especially large number were from northwestern Ontario or were native Indians (primarily Ojibwa). Bone was the second most common site of involvement (seventeen patients); in approximately one-half of these patients, the osseous site did not cause symptoms. Metaphyses of long bones and small bones were involved the most; the metaphyseal lesions tended to be eccentric, well circumscribed, and lytic. There were two deaths--the most recent, more than twenty years ago--of patients who had involvement of bones and joints; these patients, by modern standards, had had inadequate treatment. Treatment with amphotericin B and, more recently, with ketoconazole, in conjunction with operative treatment, was very effective.