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罕见的狼疮性利什曼病病例,十年未确诊。

A rare case of lupoid leishmaniasis defying diagnosis for a decade.

机构信息

Department of Dermatology, Yale University, School of Medicine, New Haven, Connecticut, USA.

Section of Infectious Disease, Yale University, School of Medicine, New Haven, Connecticut, USA.

出版信息

J Cutan Pathol. 2020 Nov;47(11):1054-1057. doi: 10.1111/cup.13793. Epub 2020 Sep 6.

DOI:10.1111/cup.13793
PMID:32623733
Abstract

Cutaneous leishmaniasis (CL) is a common disease affecting millions in endemic areas worldwide. We present a case of lupoid leishmaniasis, a rare variant of CL, which clinically mimicked sarcoidosis and/or granulomatous rosacea for 10 years until ultimate diagnosis. An 82-year-old U.S. citizen with an extensive travel history presented with a 10-year history of facial plaques on the cheeks and was previously diagnosed and treated as sarcoidosis. Multiple biopsies (previously and at presentation) revealed tuberculoid granulomas with negative special stains for microorganisms and negative sterile tissue cultures for acid-fast bacilli, bacteria, and fungal organisms. A diagnosis of granulomatous rosacea was rendered and multiple medical therapies were attempted, none with sustained improvement. Repeat biopsy of a new lesion revealed intracellular organisms consistent with leishmaniasis, which was confirmed by polymerase chain reaction (PCR). Lupoid leishmaniasis is a rare presentation of CL including facial plaques that can mimic granulomatous diseases affecting the face including sarcoidosis and granulomatous rosacea. CL can sometimes be challenging to diagnose through standard histopathologic examination; immunohistochemistry for CD1a can be used to augment tissue-based examination and PCR should be sent early in cases with sufficient concern.

摘要

皮肤利什曼病(CL)是一种常见疾病,影响着全球流行地区的数百万人。我们报告了一例狼疮样利什曼病,这是 CL 的一种罕见变异,其临床表现为 10 年的类肉瘤病和/或肉芽肿性酒渣鼻,直到最终诊断。一位 82 岁的美国公民,有广泛的旅行史,表现为面颊部有 10 年的面部斑块病史,之前被诊断为并接受了类肉瘤病的治疗。多次活检(以前和现在)显示结核样肉芽肿,特殊微生物染色阴性,无菌组织培养物未检出抗酸杆菌、细菌和真菌。诊断为肉芽肿性酒渣鼻,并尝试了多种药物治疗,但均无持续改善。新病变的重复活检显示符合利什曼病的细胞内生物体,聚合酶链反应(PCR)证实了这一点。狼疮样利什曼病是 CL 的一种罕见表现,包括面部斑块,可模仿影响面部的肉芽肿性疾病,包括类肉瘤病和肉芽肿性酒渣鼻。CL 通过标准组织病理学检查有时难以诊断;CD1a 的免疫组织化学可用于增强基于组织的检查,对于有足够关注的病例,应早期进行 PCR 检测。

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