Disseminated Basidiobolomycosis in an Immunocompetent Woman

Abstract

Basidiobolomycosis is a chronic subcutaneous infection of the trunk and limbs due to Basidiobolus ranarum.The disease is well known in tropical areas, although recent cases of gastrointestinal basidiobolomycosis have also been reported in Arizona. We describe a young immunocompetent women who had presented with eosinophilia and lung infiltrates. She subsequently died, and diagnosis of disseminated basidiobolomycosis was made on the basis of histological features at autopsy. CASE REPORTA nonsmoking, 40-year-old housewife was admitted to the hospital with sudden acute dyspnea and hypotension; she died a few minutes after admission to the emergency room due to cardiorespiratory arrest.Three days before, the patient noted the appearance of shortness of breath; hard subcutaneous and painless swellings localized to the back of the shoulders and arms were also noted. For 4 months she had been monitored as an outpatient with low-grade fever and increasing eosinophilia (white cell counts were 13.800 and then 28.280; eosinophils, 11% and then 30%), an elevated erythrocyte sedimentation rate (43 mm/h) and C-reactive protein (3.9 mg/dl), and increased immunoglobulin E (2,583 kU/liter). At that time, pulmonary function tests documented airflow obstruction and chest radiographs revealed a right upper lobe infiltrate. The oxygen saturation was 98% while on room air. Laboratory tests, including percentage of lymphocytes and total count of peripheral blood, hepatitis B virus and hepatitis C virus serological markers, and human immunodeficiency virus status, didn't reveal any form of immunosuppression. She did not suffer from diabetes mellitus or peptic ulcer disease. Symptoms persisted despite antibiotics (macrolide, penicillin-cephalosporin), inhaled corticosteroids, and long-acting ␤2 agonist treatment.A high-resolution computed tomography showed patchy subpleural infiltrates with a halo sign in the right lung (Fig. 1). Enlarged mediastinal lymph nodes, right pleural effusion, and two cysts in the liver were also detected. Bronchoscopy documented a nodular submucosal infiltration involving the distal trachea and proximal right bronchial system.Mucosal biopsies from the right main bronchus showed only eosinophil infiltration and necrosis. There were no organisms, granulomas, or evidence of neoplasm. Subsequently a surgical lung biopsy was taken from the middle right lobe. The histologic findings were similar to those of the transbronchial biopsy: eosinophil infiltration and necrosis with no organism identified. Skin biopsy from subcutaneous nodules, performed 1 day before death, showed eosinophilic panniculitis, in which scattered hyphal elements were present.Autopsy report. The macroscopic examination revealed necrotic small white-yellow opacities (3 to 4 mm in diameter) in the pericardium, parietal pleura of the right hemithorax, visceral surface of the right lung, liver, spleen, kidneys, adrenal glands, pancreas, uterus and peritoneum; the stomach presented fungating necrotic mucosal lesions. Small yellowish nonulcerated nod...

Gastrointestinal basidiobolomycosis: an emerging fungal infection causing bowel perforation in a child

“…In the cutaneous form, the portal of entry is believed to be through the skin, after insect bites, scratches and minor cuts. It is most common in young children as a disease of the skin and subcutaneous tissues, involving the thighs and buttocks (Bigliazzi et al, 2004). Most cases have been reported in tropical and subtropical climates, mainly in Indonesia and East and West Africa (Antonelli et al, 1987;de Leó n-Bojorge et al, 1988;Bittencourt et al, 1991;Scholtens & Harrison, 1994;Sugar, 2005).…”

Gastrointestinal basidiobolomycosis, an emerging infection in the immunocompetent host: a report of 14 patients

“…Rarely, the genus Basidiobolus of the order Entomophthorales produces deeply invasive fungal infections (Wasim Yusuf et al, 2003). Basidiobolus ranarum is an environmental saprophyte found worldwide and isolated from decaying vegetation, foodstuff, fruits and soil, as well as from the gastrointestinal tracts of reptiles, amphibians, fish and insectivorous bats (Bigliazzi et al, 2004).…”

Gastrointestinal basidiobolomycosis in children: an overlooked emerging infection?

“…Gastrointestinal (GIB) (Hussein et al, 2007), retroperitoneal (Finelli et al, 1997;Nazir et al, 1997) and pulmonary (Bigliazzi et al, 2004) basidiobolomycosis have been reported in the medical literature. Surgery and prolonged antifungal therapy are usually required for treatment (van den Berk et al, 2006).…”