Three new cases of systemic mycosis due to Trichosporon cufaneum are reported and compared with the 23 previous reports. Two patients had acute leukemia and one patient had a lymphoblastic lymphoma. Blood cultures in two patients and cerebrospinal fluid in the third patient were positive for T. cufaneum. Only one patient recovered after antimycotic therapy and concomittent remission of his leukemia. At autopsy, the two other patients showed widespread infection with T. cufaneum. The authors conclude that diagnosis and management of such infection in the immunosuppressed host are difficult and the prognosis is poor. We describe three new cases of disseminated T. cutaneum. Of special interest are the apparent cure with amphotericin B in one patient, and, in another, meningeal involvement not previously reported.
Case Reports
Case IA 5 I-year-old white man was admitted to the intensive care unit in June 1984 for a medullary relapse of lymphoblastic lymphoma. He was treated with cytosine arabinoside, vincristine, rubidazone, prednisone, and weekly intrathecal injections of methotrexate (for prophylactic purposes). Before these injections, the patient's skin was extensively disinfected with iodinated polyridone. Four hundred mg of ketoconazole orally per day were administrated routinely. On the sixth day of hospitalization, an initial episode of fever was easily controlled with broad spectrum antibiotic therapy. Blood and urine cultures were negative. A second episode of fever occurred on the 27th day of hospitalization. Despite a change in antibiotic therapy and parented administration of antifungal drugs (4 g/d 5-fluorocytosine and gradually increasing doses of amphotencin B, to a maximum of 0.5 mg/kg/d) the patient remained febrile. Repeated leukocyte transfusions (2 X lo8 cells/kg/d) transiently brought down his temperature. The stool examination revealed a few colonies of Candida albicans, without any evidence of systemic infection with candida. The cerebrospinal fluid (CSF), sampled on the 40th day of hospitalization during the last intrathecal injection, was clear and the culture negative, with normal protein, glucose, and chloride levels.Ten days after this last injection, the patient exhibited a sudden decrease in mental status, urinary retention and fecal incontinence, but no meningeal signs. Lumbar puncture produced a turbid liquid containing 560 leukocytes per mm3 (90% polymorphonuclear cells) and 3.5 g/l of protein (normal level, <0.35 g/l). Glucose (2.5 mmoles per 9 mmoles of serum glucose) and chloride levels (85 mmoles; normal level, I 10 mmoles) were abnormally low. CSF cultures were positive for T. cutaneum. No point source was found in fungal examination of isolates from the nose, mouth, throat, skin, urine, and stool. In addition, blood cultures were negative, as were the subclavian catheter tip culture and bone marrow culture.The patient was treated with 1.2 g/d ketoconazole orally, 6 g/d fluorocytosine and 1 mg/kg/d amphotericin B intravenously (IV). The patient was also given intrathecal inject...