Department of Laboratory Medicine and Pathology, School of Medicine, University of Washington, Seattle, USA.
Department of Pathology, University of Utah, Salt Lake City, USA.
Mycopathologia. 2022 Dec;187(5-6):605-610. doi: 10.1007/s11046-022-00654-5. Epub 2022 Aug 9.
Isolation of Cokeromyces recurvatus, a dimorphic mucormycete fungus, from clinical specimens poses a diagnostic challenge to physicians and laboratorians as this organism may represent a rare colonizer or true pathogen. Here, we report a case of Cokeromyces recurvatus present in a circumferential duodenal lesion. The patient is a 64-year-old with no past medical history, admitted with a three-week history of left upper quadrant abdominal pain. Computerized tomography scan identified duodenitis with significant gastric outlet obstruction, confirmed by the presence of a partially obstructing non-bleeding duodenal ulcer on upper endoscopy. Histology showed variably sized spherical structures without nuclei, reproductive tracts, or alimentary tracts. Small, clustered spherules representing putative endospores were observed within the larger structures and in the exudate. Based on the histology, the differential included Coccidioides spp, Emmonsia spp, or Chrysosporium spp. Additionally, gastric biopsies revealed concurrent Helicobacter pylori gastritis. The fungus was identified as C. recurvatus by broad-range fungal polymerase chain reaction performed on formalin-fixed paraffin-embedded biopsy tissue, as well as morphology and DNA sequencing of the cultured isolate. The fungus had low MICs to all major antifungal classes; however, in the context of the Helicobacter pylori infection, the patient was only treated with amoxicillin and clarithromycin with improvement in his symptoms before hospital discharge. Only three cases of Cokeromyces recurvatus isolated from the GI tract have been reported; this case highlights a unique clinical presentation in the small bowel in a patient without underlying medical conditions.
从临床标本中分离出卷曲毛霉(Cokeromyces recurvatus),这种二相性毛霉真菌,对医生和实验室人员来说是一个诊断挑战,因为这种生物体可能代表罕见的定植菌或真正的病原体。在这里,我们报告了一例卷曲毛霉存在于环状十二指肠病变中的病例。患者为 64 岁,无既往病史,因左上腹疼痛三周入院。计算机断层扫描发现有胃炎伴胃出口梗阻,上内窥镜检查发现部分阻塞性非出血性十二指肠溃疡证实了这一点。组织学显示大小不一的球形结构,无核、生殖道或消化道。在较大的结构内和渗出物中观察到小的、簇状的小球体,代表推测的内生孢子。根据组织学,鉴别诊断包括球孢子菌属、埃姆斯菌属或 Chrysosporium spp。此外,胃活检显示同时存在幽门螺杆菌胃炎。通过对福尔马林固定石蜡包埋活检组织进行广谱真菌聚合酶链反应,以及对培养分离株的形态和 DNA 测序,鉴定该真菌为卷曲毛霉。该真菌对所有主要抗真菌类别的 MIC 均较低;然而,在幽门螺杆菌感染的情况下,该患者仅接受阿莫西林和克拉霉素治疗,在出院前症状有所改善。仅从胃肠道分离出三例卷曲毛霉;该病例突出了一种在无潜在疾病的患者中小肠的独特临床表现。