Zygomycosis: Two case reports and review of reported cases in the literature in Japan

Abstract

This article reports two cases of zygomycosis and analyzes the zygomycosis cases reported in the literature in Japan. Case 1 was a 43-year-old male with malignant lymphoma who presented complications of pneumonia and cerebral bleeding, leading to his death. Autopsy findings showed pulmonary lesions were due to zygomycosis. Cerebral lesion was presumed to be due to zygomycosis without pathological examination. Case 2 was a 52-year-old male with acute lymphocytic leukemia from whom 4 sputum cultures were taken that were positive for Cunninghamella elegans. Combination therapy of itraconazole and amphotericin B (AMPH) was begun, and AMPH was changed to liposomal amphotericin B. During the neutropenic period after receiving premedication for a peripheral blood stem cell transplantation performed for his underlying disease, high fever was recognized and Staphylococcus epidermidis was isolated from the blood culture. Despite the change in antibiotics administered, pneumonia also developed as a complication, causing his death. Two hundred four cases of zygomycosis have been reported in the literature in Japan: 55 cases were rhinocerebral zygomycosis, including 29 cases with no underlying disease. A premortem diagnosis was made in 34 cases by pathological findings of operation materials or drainage samples, and 24 cases were postmortem. Pulmonary, disseminated, cardiovascular, gastrointestinal and thyroidal zygomycoses were found in 144 cases, including 66 cases with leukemia. A premortem diagnosis was made in 39 cases and 120 cases were postmortem. Prognosis of rhinocerebral type was better in operated or drainage cases, and for resected cases in all other types. Five cases with allergic zygomycosis were all alive. There were only 14 cases in which isolated fungi were identified (Cunninghamella spp. from 5 cases, Mucor spp. from 2, Rhizomucor spp. from 2, and Rhizopus spp. from 5).