Femoral head aseptic necrosis is a common complication after HSCT. In allogeneic HSCT recipients, hip tuberculosis on top of aseptic necrosis is infrequent and the mortality is high. We present a case of hip joint tuberculosis in a 57-year-old man with acute myelomonocytic leukemia (M4) treated with HSCT. The patient developed extensive chronic graft versus host disease (cGVHD) five months after transplantation and was treated with cyclosporine and corticosteroids. Eight months after the transplantation because of low-grade fever, elevated ESR and abnormal chest CT scan findings, empirical anti-TB treatment started despite negative tuberculin skin test. Three weeks later anti-TB treatment was stopped because of hepatic enzyme elevation. One year after the transplantation he complained about bilateral hip pain. MRI revealed bilateral femoral head aseptic necrosis. One year later, the right femoral head collapsed, and suddenly, rapid hip joint destruction occurred. He was planned to have total right hip arthroplasty. During the operation an abscess was evacuated and biopsy showed tuberculosis. Necrotic tissues and bone were removed and suction drainage was applied. Diagnosis was confirmed by acid-fast stain, PCR and cultures. In BACTEC MGIT 960 culture system and on Löwenstein-Jensen Mycobacterium tuberculosis was isolated, which was sensitive to all first line anti-TB drugs. After one year of anti-TB treatment (HRZE for 2 months followed by HRE for 10 months), synovial fluid samples were negative for tuberculosis. The patient was submitted to cementless total left hip replacement. Three months later, the right hip was allografted on the acetabular side and a reinforcement ring was used in order to perform a successful total hybrid arthroplasty. Nine months postoperatively the patient is symptom free and able to walk. Tuberculosis should be considered in the differential diagnosis when rapid joint destruction occurs. Early diagnosis improves response to anti-TB therapy and surgery.