A 17 years old male patient referring to a private medical clinic with an arthritis at his left knee was given non steroidal anti-inflammatory therapy. As his arthritis continued, he was admitted to our hospital. In 1988, he had a craniotomy and a cerebral mass was excised but he had no further pathology reports concerning the excised material and he was continuing to take antiepileptic medications. In 1994 for pulmonary tbc, he had been given antituberculosis therapy for one year. He had no trauma or surgical intervention to his left knee. In his physical examination, his body temperature was 39.2 C, multiple lymphadenopathy with different sizes at the axillar region, hepatosplenomegaly and arthritis at his left knee was detected. Laboratory examination revealed erythrocyte sedimentation rate (ESR) 86 mm/h, hemoglobin 9.5 gr/dl, leukocyte 5900 /mm3, thrombocyte 473 000/m3, CRP 221 mg/dl (normal < 5 mg/dl), Ig G 20.9 g/l (normal 7-16 g/l), Ig A 5.5 g/l (normal 0.7-4 g/l), Ig M 2.8 g/l (normal 0.4-2.3 g/l), albumin 24 mg/dl (normal > 35 mg/dl), PPD 20 mm diameter and HIV, rheumatoid factor and brucella agglutination tests were all negative. All the other biochemical tests were normal. X rays and magnetic resonance imaging (MRI) of the left joint showed septic arthritis (Figure 1) and patellar osteomyelitis (Figure 2). As the synovial fluid analysis displayed 80 % polymorphonuclear leukocytes, ceftriaxone 2 g/day and teicoplanin 400 mg/day were intitiated with the diagnosis of septic arthritis. His cranial MRI was normal except secondary findings due to his previous cranial operation. In the computerized tomography (CT) of the thoracal region, there were multiple lymph nodes at the axillary region and multiple calcified lymph nodes at the hilar region but bilateral pulmonary paranchimal regions were normal. The results of the blood cultures were negative. At the examination of synovial fluid, AARB was negative. Aspergillus fumigatus was isolated from the synovial fluid. Excisionel biopsies were taken from the left knee synovium and axillary lymph node. Histopathological examination of the left knee specimen expressed granulomatous lesion due to Aspergillus (Figure 3) and the lymph node biopsy from the axillary region showed changes compatible with the granulomatous disease. The patellar osteomyelitic tissue was surgically excised and another synovial fluid sample was taken from the left knee at the same session. From the tissue samples cultures Aspergillus fumigatus were yielded. Antibiotic treatment switched to Amphotericin B deoxicolate 0.7 mg/kg/day. G6PD level were normal. As the nitroblue tetrazolium test (NBT) was positive, the patient was diagnosed as chronic granulomatous disease. Surgical debridement was performed. On the 37 th day at the total dosage of 820 mg of amphotericin B nephrotoxicity developed and the therapy switched to itraconazole 400 mg/day. The infection was treated with surgical debridement and antifungal treatment. At the 37th day, ESR decreased to 25 mm/h and CRP was <5 mg/dl. The surgical wound healing was very slow and we could not introduce IFN-gamma because the licensed use of this preparation is not available in our country. The patient was given co-trimoxazole for prophylaxis and itraconazole therapy. As the surgical wound healed, the co-trimoxazole prophylaxis and itraconazole therapy were discontinued at the 6th month. The limitation of the range of motion of the knee joint excellently responded to physical therapy and rehabilitation program that lasted three weeks and a knee joint flexion of 90 degrees was achieved. The patient can perform all the activities of daily living including the school activities and he is not receiving any prophylactic medicine.