Fusarium species are representative of the emerging group of filamentous molds, which cause respiratory and disseminated infections in immunocompromised patients. To date, only five cases of respiratory or disseminated skin infections due to Fusarium spp. have been described in Korea. Here we describe a fungemia case of Fusarium oxysporum in a 3-year old boy who was neutropenic following chemotheray for leukemia. Fever, painful macules on both extremities and phlebitis on the site of venous blood sampling developed on the day 35 of admission. All four blood cultures obtained on hospital days 37, 38, 40 and 42 yielded the same F. oxysporum. The infection was cured with a high dose (1.5 mg/kg) of amphotericin B. This case shows that Fusarium is among a few filamentous fungi that cause clinically detectable fungemias in immuncompromised hosts. (Korean J Clin Microbiol 2005;8(2):189-193)