Mycobacterium szulgai is a rare cause of human infections, and when present it is mostly known to cause lung infection. We report the first case of isolated Mycobacterium szulgai osteomyelitis in a 68-year-old woman on chronic immunosuppressive therapy. Review of the literature revealed a total of three other cases of M. szulgai osteomyelitis. In all these cases there was evidence of hematogenous or contiguous spread, including one with extensive dissemination. Pulmonary M. szulgai infections tend to occur in patients with chronic lung disease and/or concomitant lung infections, whereas osteomyelitis tends to occur in patients who have severe immunosuppression secondary to disease or drugs.