From December 1996 through September 1997, we diagnosed 19 cases of fungemia due to Exophiala jeanselmei. We conducted a matched case-control study in which we cultured specimens of blood products, intravenous solutions, and water from a hospital water system. Isolates from environmental cultures were compared to those recovered from patients by random amplification of polymorphic DNA (RAPD). Multivariate analysis showed that neutropenia, longer duration of hospitalization, and use of corticosteroids were risk factors for infection. Environmental cultures yielded E. jeanselmei from 3 of 85 sources: deionized water from the hospital pharmacy, 1 water tank, and water from a sink in a non-patient care area. Use of deionized pharmacy water to prepare antiseptic solutions was discontinued, and no additional cases of infection occurred. RAPD typing showed that isolates from case patients and isolates from the pharmacy water were highly related, whereas the patterns of isolates recovered from the 2 other sources of water were distinct.