To assess the spectrum and outcome of trichosporonosis (TS) in cancer patients, we reviewed the medical records of 17 such patients with TS. TS presented most commonly as fungemia (n = 10, including 7 with central-venous-catheter-related infection) and either pulmonary or soft tissue infection (n = 3, each). Most patients (65%) had acute leukemia, 11 (65%) had neutropenia, and 9 (53%) had received high doses of corticosteroids. 10 patients had breakthrough TS during therapy with at least 1 of the following: amphotericin B, fluconazole, itraconazole, and voriconazole. The 30-d crude mortality rate was 53%. Predictors of mortality by using univariate analysis included: high median APACHE II score (p < 0.01), use of high dose of corticosteroids (p = 0.01), and admission to the intensive care unit (p < 0.01). TS is associated with a high mortality rate in cancer patients. The spectrum of infection at our institution has shifted from a predominance of disseminated infection to CVC-related fungemias without evidence of tissue invasion.