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Case Reports in Pathology
Volume 2018, Article ID 1351694, 6 pages
Case Report

Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin

1Department of Pathology, School of Medicine, University of Parakou, 03 BP 333 Parakou, Benin
2Department of Pathology, Hospital of Chambéry, Place Lucien Biset, 73000 Chambéry, France
3Buruli Ulcer Treatment Center, Allada, Benin
4Regional Institute of Public Health, Ouidah, Benin
5Nutritional Center of Gbemontin, Zagnanado, Benin
6Institute of Tropical Medicine, Nationalestraat 155, 2000 Antwerpen, Belgium
7Armed Forces Institute of Pathology, Washington, DC, USA
8BCCM/IHEM Biomedical Fungi and Yeasts Collection, Scientific Institute of Public Health, Brussels, Belgium
9Department of Pathology, School of Medicine, University of Abomey Calavi, Cotonou, Benin

Correspondence should be addressed to Luc V. C. Brun; moc.liamg@3102nurb.cul

Received 11 August 2017; Accepted 11 December 2017; Published 10 January 2018

Academic Editor: Mark Li-cheng Wu

Copyright © 2018 Luc V. C. Brun et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.


Background. Basidiobolomycosis is a rare subcutaneous mycosis, which can be mistaken for several other diseases, such as soft tissue tumors, lymphoma, or Buruli ulcer in the preulcerative stage. Microbiological confirmation by PCR for Basidiobolus ranarum and culture yield the most specific diagnosis, yet they are not widely available in endemic areas and with varying sensitivity. A combination of histopathological findings, namely, granulomatous inflammation with giant cells, septate hyphal fragments, and the Splendore-Hoeppli phenomenon, can confirm basidiobolomycosis in patients presenting with painless, hard induration of soft tissue. Case Presentations. We report on three patients misdiagnosed as suffering from Buruli ulcer, who did not respond to Buruli treatment. Histopathological review of the tissue sections from these patients suggests basidiobolomycosis. All patients had been lost to follow-up, and none received antifungal therapy. On visiting the patients at their homes, two were reported to have died of unknown causes. The third patient was found alive and well and had experienced local spontaneous healing. Conclusion. Basidiobolomycosis is a rare subcutaneous fungal disease mimicking preulcerative Buruli ulcer. We stress the importance of the early recognition by clinicians and pathologists of this treatable disease, so patients can timely receive antifungal therapy.