Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin
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 diagnosi...
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Wiley
2018-01-01
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| Series: | Case Reports in Pathology |
| Online Access: | http://dx.doi.org/10.1155/2018/1351694 |
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| author | Luc V. C. Brun Jean Jacques Roux Ghislain E. Sopoh Julia Aguiar Miriam Eddyani Wayne M. Meyers Dirk Stubbe Marie T. Akele Akpo Françoise Portaels Bouke C. de Jong |
| author_facet | Luc V. C. Brun Jean Jacques Roux Ghislain E. Sopoh Julia Aguiar Miriam Eddyani Wayne M. Meyers Dirk Stubbe Marie T. Akele Akpo Françoise Portaels Bouke C. de Jong |
| author_sort | Luc V. C. Brun |
| collection | DOAJ |
| description | 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. |
| format | Article |
| id | doaj-art-f0e00816c0a44bdc8d7787699d25221b |
| institution | Kabale University |
| issn | 2090-6781 2090-679X |
| language | English |
| publishDate | 2018-01-01 |
| publisher | Wiley |
| record_format | Article |
| series | Case Reports in Pathology |
| spelling | doaj-art-f0e00816c0a44bdc8d7787699d25221b2025-02-03T01:23:22ZengWileyCase Reports in Pathology2090-67812090-679X2018-01-01201810.1155/2018/13516941351694Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in BeninLuc V. C. Brun0Jean Jacques Roux1Ghislain E. Sopoh2Julia Aguiar3Miriam Eddyani4Wayne M. Meyers5Dirk Stubbe6Marie T. Akele Akpo7Françoise Portaels8Bouke C. de Jong9Department of Pathology, School of Medicine, University of Parakou, 03 BP 333 Parakou, BeninDepartment of Pathology, Hospital of Chambéry, Place Lucien Biset, 73000 Chambéry, FranceBuruli Ulcer Treatment Center, Allada, BeninNutritional Center of Gbemontin, Zagnanado, BeninInstitute of Tropical Medicine, Nationalestraat 155, 2000 Antwerpen, BelgiumArmed Forces Institute of Pathology, Washington, DC, USABCCM/IHEM Biomedical Fungi and Yeasts Collection, Scientific Institute of Public Health, Brussels, BelgiumDepartment of Pathology, School of Medicine, University of Abomey Calavi, Cotonou, BeninInstitute of Tropical Medicine, Nationalestraat 155, 2000 Antwerpen, BelgiumInstitute of Tropical Medicine, Nationalestraat 155, 2000 Antwerpen, BelgiumBackground. 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.http://dx.doi.org/10.1155/2018/1351694 |
| spellingShingle | Luc V. C. Brun Jean Jacques Roux Ghislain E. Sopoh Julia Aguiar Miriam Eddyani Wayne M. Meyers Dirk Stubbe Marie T. Akele Akpo Françoise Portaels Bouke C. de Jong Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin Case Reports in Pathology |
| title | Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin |
| title_full | Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin |
| title_fullStr | Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin |
| title_full_unstemmed | Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin |
| title_short | Subcutaneous Granulomatous Inflammation due to Basidiobolomycosis: Case Reports of 3 Patients in Buruli Ulcer Endemic Areas in Benin |
| title_sort | subcutaneous granulomatous inflammation due to basidiobolomycosis case reports of 3 patients in buruli ulcer endemic areas in benin |
| url | http://dx.doi.org/10.1155/2018/1351694 |
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