Scedosporium prolificans is a saprophytic fungus widespread in the environment. It has become an emerging pathogen in recent years causing disseminated infections, especially in profoundly neutropenic immunocompromised patients. We report a case of fatal Scedosporium fungemia in a 45 year old female with acute myeloid leukemia in relapse. She received salvage chemotherapy and antibiotic treatment, and was neutropenic with relapsing fever. S. prolificans was isolated repeatedly from the aerobic bottles on the second day of two successive blood cultures. Amphotercin B was started; however, the patient expired the next day. (Korean J Clin Microbiol 2005;8(2):194-197)