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[丙硫氧嘧啶诱导的抗蛋白酶3抗中性粒细胞胞浆抗体(PR3-ANCA)相关的坏疽性脓皮病]

[Pyoderma gangrenosum associated with anti-proteinase 3 antineutrophil cytoplasmic antibodies (PR3-ANCA) induced by propylthiouracil].

作者信息

Coster A, Dargent J-L, de Visscher N, Levecque P, Roquet-Gravy P-P

机构信息

Département de dermatologie, grand hôpital de Charleroi (GHDC), Grand'Rue 3, 6000 Charleroi, Belgique.

Département d'anatomie pathologique, institut de pathologie et de génétique (IPG), 25, avenue Georges-Lemaître, 6041 Gosselies, Belgique.

出版信息

Ann Dermatol Venereol. 2017 May;144(5):368-373. doi: 10.1016/j.annder.2017.01.018. Epub 2017 Mar 11.

Abstract

BACKGROUND

Synthetic antithyroid drugs are often used in the treatment of hyperthyroidism, regardless of aetiology. They may cause various side effects, including the development of anti-neutrophil cytoplasmic antibodies (ANCA), ANCA-associated vasculitis, and neutrophilic dermatoses. Propylthiouracil (PTU) is the antithyroid drug most frequently implicated in ANCA-associated diseases specifically involving anti-myeloperoxidase ANCA (MPO-ANCA). To our knowledge, there are no clinical reports describing the association of pyoderma gangrenosum (PG) and anti-proteinase3-ANCA (PR3-ANCA) induced by PTU, with ANCA levels decreasing after antithyroid drug withdrawal.

PATIENTS AND METHODS

A 68-year-old woman was treated with propylthiouracil (PTU) for toxic multinodular goitre. She presented necrotic ulceration of the lower abdomen. The patient's history, physical examination, and bacteriological and histological samples led to a diagnosis of pyoderma gangrenosum. This pyoderma involved ANCA with antigenic specificity for proteinase 3. Withdrawal of PTU and a short course of corticosteroids and cyclosporine resulted in rapid and complete resolution of the pyoderma gangrenosum as well as a decrease in ANCA. No relapse was observed one year after cessation of treatment.

DISCUSSION

We report a case of PG associated with PR3-ANCA induced by PTU, without any demonstrable vasculitis.

摘要

背景

无论病因如何,合成抗甲状腺药物常用于治疗甲状腺功能亢进症。它们可能会引起各种副作用,包括抗中性粒细胞胞浆抗体(ANCA)的产生、ANCA相关血管炎和嗜中性皮病。丙硫氧嘧啶(PTU)是最常与ANCA相关疾病有关的抗甲状腺药物,特别是涉及抗髓过氧化物酶ANCA(MPO-ANCA)。据我们所知,尚无临床报告描述PTU诱导的坏疽性脓皮病(PG)与抗蛋白酶3-ANCA(PR3-ANCA)之间的关联,且停用抗甲状腺药物后ANCA水平会降低。

患者和方法

一名68岁女性因毒性多结节性甲状腺肿接受丙硫氧嘧啶(PTU)治疗。她出现了下腹部坏死性溃疡。患者的病史、体格检查以及细菌学和组织学样本导致诊断为坏疽性脓皮病。这种坏疽性脓皮病涉及对蛋白酶3具有抗原特异性的ANCA。停用PTU并短期使用皮质类固醇和环孢素后,坏疽性脓皮病迅速完全消退,ANCA水平也降低。治疗停止一年后未观察到复发。

讨论

我们报告了一例由PTU诱导的与PR3-ANCA相关的PG病例,且无任何可证实的血管炎。

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