Osteonecrosis of the femoral head is rare in human immunodeficiency virus (HIV)-infected patients. We describe a 37-year-old HIV-infected man who developed lipodystrophy after highly active antiretroviral therapy (HAART), leading to left leg disability. Radiography revealed osteonecrosis of the left femoral head, which appeared to be related to the antiretroviral therapy for HIV infection and subsequent development of lipodystrophy and hyperlipidemia. While osteonecrosis among Taiwanese HIV-infected patients is uncommon, the potential for the development of this long-term complication should be carefully monitored as more patients survive for longer periods after HAART.