Trichosporon beigelii is often resistant to the fungicidal effect of amphotericin B and can cause fatal disseminated infections in immunocompromised patients. We report a case of a disseminated T. beigelii infection with a favorable outcome in a patient with acute erythroleukemia and neutropenia. The patient presented a persistent fever, multiple erythematous skin lesions, and pulmonary infiltrates. T. beigelii was isolated from blood cultures in four days and also from cultures of abdominal skin lesion, sputum, and stool. The isolate was resistant to amphotericin B (MIC, 2 μg/mL), and the respective fluconazole and itraconazole MICs were 4 and 1 μg/mL. The patient was successfully treated with fluconazole plus amphotericin B in combination with granulocyte colony stimulating factor and leukocyte transfusion. This case shows the importance of early diagnosis and treatment with combination of amphotericin B and fluconazole as a prognostic factor of disseminated T. beigelii infections. (Korean J Clin Microbiol 2005;8(2):179-184)