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浸润性、类癌性直肠病变:内脏荚膜组织胞浆菌的独特表现。

Infiltrating, Quasi-Cancerous Rectal Lesions: Unique Manifestation of Visceral Basidiobolus ranarum.

机构信息

Department of Pediatric Surgery, Abha Maternity and Children's Hospital, Abha, 62521, Saudi Arabia.

Department of Radiology, College of Medicine, King Khalid University and Abha Maternity and Children's Hospital, Abha, Saudi Arabia.

出版信息

Indian J Pediatr. 2018 Dec;85(12):1096-1100. doi: 10.1007/s12098-018-2728-7. Epub 2018 Jun 14.

DOI:10.1007/s12098-018-2728-7
PMID:29948734
Abstract

OBJECTIVE

To report the finding of transmural rectal involvement, in four children with lesions which seemed to be neoplastic in appearance and progression.

METHODS

The case records of four children presenting with rectal involvement by the fungus Basidiobolus ranarum, were retrospectively analyzed for clinical presentation, hematological and radiological investigations, the procurement of histopathological material for diagnosis and the findings thereof, the treatment of these patients and the follow-up.

RESULTS

The four children presented with non-specific symptoms of fever, loss of weight and appetite, constipation and bleeding per rectum. One presented with excoriation and ulceration of the perineum and perianal skin. Examination generally unremarkable, however, showed the rectum extensively involved by a lesion which narrowed the lumen. Hematological investigations showed leukocytosis with eosinophilia and raised levels of acute phase reactants like ESR, platelets, and C-reactive proteins. Ultrasonogram (USG) and CT scan confirmed the lesion to be involving all layers of the rectum and compromising the rectal lumen. The right colon was also involved in one patient. One underwent a colonoscopy and biopsy which proved inconclusive. All four underwent a Tru-cut biopsy which was diagnostic. Histopathologically the disease was based in Basidiobolus ranarum, a fungus which is emerging as a cause of visceral abdominal involvement.

CONCLUSIONS

Lesions involving the rectum, and appearing to be neoplastic may be caused by the fungus Basidiobolus ranarum. The symptomatology and presentation may be non-specific. High levels of eosinophils in the blood, a raised ESR, and C-reactive protein may be useful pointers to the diagnosis. USG and CT scans localize the lesions and also provide a guide for biopsies. The characteristic histopathological findings are diagnostic and based on these, treatment with Itroconazole / Voriconazole is beneficial.

摘要

目的

报告 4 例儿童患者的直肠壁受累情况,这些患者的病变外观和进展似乎为肿瘤性。

方法

回顾性分析了 4 例直肠被蛙粪霉菌(Basidiobolus ranarum)感染的患儿的病例记录,分析内容包括临床表现、血液学和影像学检查、获取组织病理学材料进行诊断及发现、这些患者的治疗方法及随访情况。

结果

4 例患儿均表现出发热、消瘦、食欲减退、便秘和直肠出血等非特异性症状。1 例患儿出现肛周皮肤皲裂和溃疡。一般体格检查无明显异常,但直肠广泛受累,导致管腔变窄。血液学检查显示白细胞增多伴嗜酸性粒细胞增多,急性反应物质如 ESR、血小板和 C-反应蛋白水平升高。超声(USG)和 CT 扫描证实病变累及直肠的所有层,使直肠管腔狭窄。1 例患者的右半结肠也受累。1 例患者进行了结肠镜检查和活检,但结果不确定。所有 4 例患者均接受了 Tru-cut 活检,结果均为阳性。组织病理学上,该病由蛙粪霉菌(Basidiobolus ranarum)引起,该真菌正成为内脏腹部感染的原因之一。

结论

累及直肠且外观似肿瘤的病变可能由蛙粪霉菌引起。症状和表现可能不具有特异性。血液中嗜酸性粒细胞水平升高、ESR 和 C-反应蛋白升高可能有助于诊断。USG 和 CT 扫描可定位病变,并为活检提供指导。特征性的组织病理学发现具有诊断意义,基于这些发现,使用伊曲康唑/伏立康唑治疗是有益的。

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Pediatric gastrointestinal basidiobolomycosis mimicking malignancy.酷似恶性肿瘤的小儿胃肠道担子菌病
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Gastrointestinal basidiobolomycosis, an emerging infection in the immunocompetent host: a report of 14 patients.胃肠道芽生菌病,一种免疫功能正常宿主中的新发感染:14 例患者报告。
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Gastrointestinal basidiobolomycosis in Arizona: clinical and epidemiological characteristics and review of the literature.亚利桑那州的胃肠道担子菌病:临床和流行病学特征及文献综述
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