We report the first case, to our knowledge, of Blastoschizomyces capitatus infection occurring in a patient receiving empirical echinocandin therapy for neutropenic fevers. Clinicians should consider B. capitatus infection in those neutropenic patients who remain febrile despite echinocandin therapy or who develop yeast bloodstream infections while receiving an echinocandin.The febrile neutropenic patient has been and continues to be a major therapeutic challenge (5). As our armamentarium of effective prophylactic and empirical treatment options has expanded, the pathogens causing infections have evolved in response to those changes. Using antimicrobial agents with good activity against particular organisms has led to an increasing prevalence of infections with organisms not typically covered by these agents. As an example of this phenomenon, we report the first known case of Blastoschizomyces capitatus infection occurring in a host receiving empirical therapy with an echinocandin.A 65-year-old Caucasian male was admitted to our institution with complaints of fatigue and shortness of breath. A complete blood count at admission revealed pancytopenia, with circulating blasts. The results of a bone marrow biopsy confirmed a diagnosis of acute myelogenous leukemia. A peripherally inserted central venous catheter (PICC) was placed in the patient, and he was treated with cytarabine and daunorubicin. After initial prophylaxis with moxifloxacin, fluconazole, and acyclovir, he became neutropenic and febrile on hospital day 8, at which time he was started on vancomycin and cefepime treatment. A coagulase-negative staphylococcus PICC-associated bloodstream infection was diagnosed, and he responded well to treatment with retention of the PICC. On hospital day 20, he again developed fevers, and fluconazole was changed to micafungin (100 mg daily). The blood cultures were negative, and the patient defervesced while on the same antimicrobial regimen. On hospital day 34, he was begun on total parenteral nutrition due to inadequate oral intake and again developed fevers. His cefepime was changed to meropenem, but his fevers persisted. Over the next 3 days, he manifested worsening renal, hepatic, and pulmonary dysfunction. Multiple blood cultures obtained on hospital days 34, 35, 36, and 37 ultimately all grew Blastoschizomyces capitatus. His PICC was removed on hospital day 35; a culture of the catheter tip was negative. On hospital day 37, the patient's micafungin was changed to amphotericin B lipid complex and intravenous voriconazole on the basis of the first set of cultures yielding positive results, but he died later that day after a cardiopulmonary arrest.