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一名患有长期黑利-黑利病并伴有疱疹样湿疹的患者出现大疱性类天疱疮:应对三种不同水疱性疾病并存的情况。

Bullous Pemphigoid in a Patient with Longstanding Hailey-Hailey Disease Complicated by Eczema Herpeticum: Managing the Coexistence of Three Different Blistering Conditions.

作者信息

Calvão Da Silva Joana Cruz Matos, Vieira Ricardo, Brites Maria Manuel

机构信息

Drs. Calvão da Silva, Vieira, and Brites are with the Dermatology Department at Coimbra University Hospital in Coimbra, Portugal.

出版信息

J Clin Aesthet Dermatol. 2020 Mar;13(3):17-19. Epub 2020 Mar 1.

PMID:32308791
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7159308/
Abstract

Hailey-Hailey disease (HHD), or chronic benign familial pemphigus, is a rare inherited acantholytic dermatosis, characterized by chronic, recurrent vesicles, erosions, and maceration in intertriginous sites. We present a case of a male patient with longstanding HHD who presented with an acute exacerbation characterized by the worsening of pre-existing lesions but also with the appearance of new large, tense bullae on an erythematous base in the areas of the groin (i.e., inguinal region), trunk, and arms, associated with intense pruritus. Blood work revealed eosinophilia. Histopathology and direct immunofluorescence were compatible with the diagnosis of bullous pemphigoid (BP). Indirect immunofluorescence showed positivity for autoantibodies to BP antigen 180. We started oral methylprednisolone, oral antihistamines, and local care with potassium permanganate baths, a potent corticosteroid, and fusidic acid, with resulting improvement of the lesions. The case was further complicated by the occurrence of eczema herpeticum, which was successfully treated with acyclovir. At the time of discharge from the hospital, the patient was medicated with a low dose of oral steroid and oral doxycycline. During a later examination, the lesions had totally disappeared, but the skin had some residual hyperpigmented patches and excoriated papules. This case was a diagnostic challenge due to the simultaneous occurrence of three distinct bullous diseases with different etiopathogeneses. To our knowledge, there are no other reports of the coexistence of HHD and BP in the literature.

摘要

黑利-黑利病(HHD),即慢性良性家族性天疱疮,是一种罕见的遗传性棘层松解性皮肤病,其特征为在皮肤褶皱部位出现慢性、复发性水疱、糜烂和浸渍。我们报告一例长期患有HHD的男性患者,其急性加重表现为原有皮损恶化,同时在腹股沟区(即腹股沟部位)、躯干和手臂出现新的、在红斑基础上的大而紧张的水疱,并伴有剧烈瘙痒。血液检查显示嗜酸性粒细胞增多。组织病理学和直接免疫荧光检查结果与大疱性类天疱疮(BP)的诊断相符。间接免疫荧光显示针对BP抗原180的自身抗体呈阳性。我们开始给予口服甲泼尼龙、口服抗组胺药,并采用高锰酸钾浴、强效皮质类固醇和夫西地酸进行局部护理,皮损有所改善。该病例因发生疱疹样湿疹而进一步复杂化,后者经阿昔洛韦成功治疗。出院时,患者服用低剂量口服类固醇和口服多西环素。在随后的检查中,皮损完全消失,但皮肤有一些残留的色素沉着斑和抓痕性丘疹。由于同时出现三种病因不同的明显大疱性疾病,该病例在诊断上具有挑战性。据我们所知,文献中尚无HHD和BP共存的其他报道。

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本文引用的文献

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RETRACTED: Bullous pemphigoid: associations and management guidelines: facts and controversies.撤回:大疱性类天疱疮:关联和管理指南:事实和争议。
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