Soleimani Neda, Anbardar Mohammad Hossein, Nikoupour Hamed, Derakhshan Faranak, Shafiekhani Mojtaba, Mohammadzadeh Sahand, Sakhaei Seyed Mohamad, Farhadi Mahsa
Department of Pathology Shiraz Medical School Shiraz University of Medical Sciences, Shiraz, Iran.
Department of Pathology Shiraz Transplant Center Abu Ali Sina Hospital Shiraz University of Medical Sciences, Shiraz, Iran.
Case Rep Med. 2024 Aug 28;2024:5741625. doi: 10.1155/2024/5741625. eCollection 2024.
Basidiobolomycosis is a rare fungal infection caused by an environmental saprophyte, . It usually presents as a chronic subcutaneous infection; however, few cases of gastrointestinal involvement have been reported. The exact transmission route of gastrointestinal cases is not clear, and diagnosis always requires a high index of suspicion because it tends to mimic other inflammatory and neoplastic conditions. . A 31-year-old immunocompetent woman presented with abdominal pain and an advanced colon mass. She was completely well until about 1.5 years ago, when she underwent bariatric surgery. One year after surgery, chronic abdominal pain developed. A colonoscopy showed an ulcerative lesion in the descending colon, and the biopsy was in favor of ulcerative colitis. Despite immunosuppressive treatment, there was no improvement, and with worsening symptoms, more investigations revealed advanced colon mass with entrapment of the stomach and pancreas. Colonic mucosa biopsy and trucut biopsy of the mass showed just necrosis and acute inflammation; thus, she underwent exploratory laparotomy with colectomy, partial gastrectomy, distal pancreatectomy, and left nephrectomy. On pathologic examination, there was granulomatous inflammation plus the Splendore-Hoeppli phenomenon around fungal hyphae, which was diagnostic for gastrointestinal basidiobolomycosis. Previous pathology slides were reviewed and revealed a tiny focus of basidiobolomycosis. After 6 months of treatment with itraconazole, she is relatively well without any clinical or radiologic abnormalities.
Our case highlights the significance of suspicion for basidiobolomycosis in ulcerative and necrotic lesions with increased eosinophils, especially in the presence of abdominal mass and systemic eosinophilia.
担子菌病是一种由环境腐生菌引起的罕见真菌感染。它通常表现为慢性皮下感染;然而,胃肠道受累的病例报道较少。胃肠道病例的确切传播途径尚不清楚,诊断总是需要高度怀疑,因为它往往会模仿其他炎症和肿瘤性疾病。一名31岁免疫功能正常的女性出现腹痛和晚期结肠肿块。直到大约1.5年前她接受减肥手术前,她一直完全健康。手术后一年,出现慢性腹痛。结肠镜检查显示降结肠有溃疡性病变,活检结果支持溃疡性结肠炎。尽管进行了免疫抑制治疗,但病情没有改善,随着症状加重,更多检查发现晚期结肠肿块并累及胃和胰腺。结肠黏膜活检和肿块的粗针活检仅显示坏死和急性炎症;因此,她接受了剖腹探查术,并行结肠切除术、部分胃切除术、远端胰腺切除术和左肾切除术。病理检查发现真菌菌丝周围有肉芽肿性炎症及Splendore-Hoeppli现象,这对胃肠道担子菌病具有诊断意义。回顾先前的病理切片发现了一个微小的担子菌病病灶。接受伊曲康唑治疗6个月后,她情况相对良好,没有任何临床或影像学异常。
我们的病例强调了对于嗜酸性粒细胞增多的溃疡性和坏死性病变,尤其是存在腹部肿块和全身嗜酸性粒细胞增多时,怀疑担子菌病的重要性。