Abstract

Cunninghamella bertholletiae, a zygomycete isolated in soil and plants in temperate climates, causes an infection with a very high mortality rate(76%). The pulmonary form is rare and occurs mostly in patients treated for leukemia. Since 1959 five cases have been reported in children (< 18-years-old), 4 with leukemia, 2 with aspergillosis co-infection and 3 deaths. A 17-year-old girl was treated for relapsed acute lymphoid leukemia. Neutropenia induced by chemotherapy was complicated by fever and was treated with a large broad spectrum antibiotic. At day 13, a chest radiograph revealed a left parahilar alveolar condensation. Direct mycological examination of the bronchoalveolar lavage sample showed mycelium very suggestive of mucorales. At the same time, the galactommanan antigen titer increased to 2.95 ng/ml. Treatment with conventional amphotericin B (Fungizone ®) was started; the patient became afebrile at the 48 th hour. After the fungal culture yielded C. bertholletiae, liposomal amphotericin B (Ambisome ®) was started. Because of the closeness of the pulmonary artery and the persistence of haemoptysis, a resection of the left upper pulmonary lobe was performed. An examination of the mycological parenchyma was negative. Microscopic examination and fungal culture of the pulmonary vein fragment were positive. Histological examination revealed mycelial invasion of the vascular and bronchial lumens and walls. Ambisome ® treatment was continued until day 57, then changed to posaconazole (Noxafil ®). On November 17 the patient received an allogenic bone marrow transplant. At the time of writing, 5 months after the transplant, she is well with no evidence of disease recurrence and posaconazole therapy is being continued.

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