Cryptococcosis is a disseminated infection of man and animals caused by Cryptococcus neoformans. The most commonly involved sites are the lungs and the central nervous system. Isolated osteomyelitis due to C. neoformans is a rare complication of disseminated cryptococcosis. Herein we report a case of isolated osteomyelitis due to C. neoformans. A review of the English-language literature has been made and shows 40 cases (including this present report) with detailed data available since 1956. Most of the cases occurred between the ages of 21 and 59. Seventy-five percent of cases involved only one single site of bone infection, with vertebrae being the most common site. Sarcoidosis is the most common underlying disease, followed by tuberculosis and previous steroid therapy. Most of the cases (> 60%) reported were treated successfully with medical treatment alone or the combination of medical treatment and surgical curettage.