Humeral osteomyelitis caused by Mycobacterium tuberculosis is rare. Since the advent of modern diagnostic techniques and antituberculous therapy, only a limited number of cases have been reported in the literature. Most patients are relatively young, free of underlying disease and live in rural areas. They always present with humeral pain and swelling. Diagnosis is based on histological examination or mycobacterial culture of infected tissue. Most patients recover after surgical debridement combined with chemotherapy. The possibility of humeral tuberculous osteomyelitis should be considered in a patient presenting with humeral pain and swelling. We report on a 6-month-old infant with tuberculosis osteomyelitis of the left proximal humerus and discuss this rare entity.