Case Report Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin Luc V. C. Brun , 1 Jean Jacques Roux, 2 Ghislain E. Sopoh , 3,4 Julia Aguiar, 5 Miriam Eddyani, 6 Wayne M. Meyers, 7 Dirk Stubbe, 8 Marie T. Akele Akpo, 9 Françoise Portaels, 6 and Bouke C. de Jong 6 1 Department of Pathology, School of Medicine, University of Parakou, 03 BP 333 Parakou, Benin 2 Department of Pathology, Hospital of Chamb´ ery, Place Lucien Biset, 73000 Chamb´ ery, France 3 Buruli Ulcer Treatment Center, Allada, Benin 4 Regional Institute of Public Health, Ouidah, Benin 5 Nutritional Center of Gbemontin, Zagnanado, Benin 6 Institute of Tropical Medicine, Nationalestraat 155, 2000 Antwerpen, Belgium 7 Armed Forces Institute of Pathology, Washington, DC, USA 8 BCCM/IHEM Biomedical Fungi and Yeasts Collection, Scientifc Institute of Public Health, Brussels, Belgium 9 Department of Pathology, School of Medicine, University of Abomey Calavi, Cotonou, Benin Correspondence should be addressed to Luc V. C. Brun; luc.brun2013@gmail.com 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. Tis 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 sof tissue tumors, lymphoma, or Buruli ulcer in the preulcerative stage. Microbiological confrmation by PCR for Basidiobolus ranarum and culture yield the most specifc diagnosis, yet they are not widely available in endemic areas and with varying sensitivity. A combination of histopathological fndings, namely, granulomatous infammation with giant cells, septate hyphal fragments, and the Splendore-Hoeppli phenomenon, can confrm basidiobolomycosis in patients presenting with painless, hard induration of sof tissue. Case Presentations. We report on three patients misdiagnosed as sufering 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. Te 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. 1. Introduction Basidiobolomycosis is a subcutaneous mycosis that occurs in tropical and subtropical regions of Asia, Africa, and South America, caused by Basidiobolus ranarum Eidam or related species [1]. Te disease mainly afects children and tends to preferentially afect the thighs, buttocks, or trunks. Te clinical and histopathological signs at frst do not evoke a fungal disease. Te initial infection is subacute, sometimes marked by a pseudophlegmon or erysipelas. Te evolution is slow and gradual, with periods of remissions over several months or years, in the form of an extensive plaque. Patients on immunosuppressive therapy may be at higher risk of developing progressive disease [2]. Clinical examination reveals inhomogeneous swellings. Palpation reveals irregular frm nodules. Te edema is nonpitting and hard as wood, and the skin is rigid and cannot wrinkle. Te symptomatology varies, depending on the evolutionary stage (early versus Hindawi Case Reports in Pathology Volume 2018, Article ID 1351694, 6 pages https://doi.org/10.1155/2018/1351694