Brun Luc V C, Roux Jean Jacques, Sopoh Ghislain E, Aguiar Julia, Eddyani Miriam, Meyers Wayne M, Stubbe Dirk, Akele Akpo Marie T, Portaels Françoise, de Jong Bouke C
Department of Pathology, School of Medicine, University of Parakou, 03 BP 333 Parakou, Benin.
Department of Pathology, Hospital of Chambéry, Place Lucien Biset, 73000 Chambéry, France.
Case Rep Pathol. 2018 Jan 10;2018:1351694. doi: 10.1155/2018/1351694. eCollection 2018.
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 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.
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.
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.
担子菌病是一种罕见的皮下真菌病，在溃疡前期可被误诊为其他多种疾病，如软组织肿瘤、淋巴瘤或布氏菌病。通过聚合酶链反应（PCR）和培养进行微生物学确诊可获得最具特异性的诊断，但在流行地区这些方法并不普遍，且敏感性各异。组织病理学表现，即伴有巨细胞的肉芽肿性炎症、分隔的菌丝片段和斯普伦多雷 - 赫普利现象，可在软组织出现无痛性硬结的患者中确诊担子菌病。
我们报告了三名被误诊为布氏菌病的患者，他们对布氏菌病治疗无反应。对这些患者组织切片的组织病理学复查提示为担子菌病。所有患者均失访，且均未接受抗真菌治疗。在到患者家中探访时，据报告有两名患者死因不明。第三名患者被发现还活着且情况良好，局部已自行愈合。
担子菌病是一种罕见的皮下真菌病，可类似布氏菌病溃疡前期表现。我们强调临床医生和病理学家早期识别这种可治疗疾病的重要性，以便患者能及时接受抗真菌治疗。
