This case report describes an unusual case of extrapulmonary tuberculosis of the ischial tuberosity presenting with chronic gluteal pain of 4 years duration. The patient presented when the pain became intolerable; at that time, the ischial tuberosity was debrided and curetted while incision and drainage were used to treat the gluteal abscess. Antituberculosis chemotherapy was administered (for a period of one year) following histopathological confirmation of tuberculosis. At four yearspostoperatively, the patient has no pain and is symptom free. Furthermore, radiographs shows that the right ischial tuberosity osteomyelitis has healed. Prompt diagnosis and treatment resulted in a good clinical outcome in this patient.