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A case of catheter-associated fungemia due to Exophiala oligosperma in a 3-year-old leukemic child is presented. The etiologic agent was isolated from blood specimens and the catheter tip. The isolate was identified by its morphological characteristics and DNA sequencing of the internal transcribed spacer region of rDNA. Despite initial amphotericin B and itraconazole therapy, the child's fever subsided only after removal of the catheter. A review of the medical literature revealed 29 cases of infection due to Exophiala species. Twenty-three of these 29 patients had a CVC in place when they developed fever or other manifestations of fungemia. Withdrawal of the CVC together with amphotericin B and/or itraconazole therapy generally resulted in a good prognosis.