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通过斑贴试验阳性确诊的丙硫氧嘧啶诱发的抗中性粒细胞胞浆抗体(ANCA)阳性皮肤白细胞破碎性血管炎:一例报告并文献复习

Antineutrophil cytoplasmic antibody (ANCA)-positive cutaneous leukocytoclastic vasculitis induced by propylthiouracil confirmed by positive patch test: a case report and review of the literature.

作者信息

Morais Paulo, Baudrier Teresa, Mota Alberto, Cunha Ana Paula, Alves Marta, Neves Celestino, Capela João, Sá-Couto Pedro, Azevedo Filomena

机构信息

Department of Dermatovenereology, Hospital São João, Alameda Professor Hernâni Monteiro, Porto, Portugal.

出版信息

Cutan Ocul Toxicol. 2011 Jun;30(2):147-53. doi: 10.3109/15569527.2010.533318. Epub 2010 Nov 24.

Abstract

A 43-year-old female with antiphospholipid syndrome and Graves' disease developed a cutaneous leukocytoclastic vasculitis associated with antineutrophil cytoplasmic antibody (ANCA) against myeloperoxidase (MPO-ANCA) and proteinase-3 (PR3-ANCA), whilst treated with propylthiouracil (PTU). The skin lesions were progressively resolved after withdrawal of PTU and treatment with oral steroids. Patch testing with PTU at 1%, 5%, and 10% in petrolatum was positive at 48 h. Despite positive ANCA titers after 1 year of follow-up, the patient maintains complete clinical remission. PTU is a common antithyroid drug, which has been known to induce ANCA-positive vasculitis. Although most patients with this rare side effect have a good outcome, some fatal cases have been reported. Therefore, patients treated with PTU should be carefully followed and monitored, not only for their thyroid state but also for early detection of potential serious complications of this drug. Early diagnosis and prompt cessation of PTU therapy are essential to improve the outcome. Also key aspects of PTU-induced ANCA-positive vasculitis are reviewed.

摘要

一名患有抗磷脂综合征和格雷夫斯病的43岁女性,在接受丙硫氧嘧啶(PTU)治疗期间,出现了与抗髓过氧化物酶抗中性粒细胞胞浆抗体(MPO - ANCA)和蛋白酶3(PR3 - ANCA)相关的皮肤白细胞破碎性血管炎。停用PTU并给予口服类固醇治疗后,皮肤病变逐渐消退。用含1%、5%和10% PTU的凡士林进行斑贴试验,48小时时呈阳性。尽管随访1年后ANCA滴度仍为阳性,但患者保持完全临床缓解。PTU是一种常见的抗甲状腺药物,已知可诱发ANCA阳性血管炎。虽然大多数有这种罕见副作用的患者预后良好,但也有一些致命病例的报道。因此,接受PTU治疗的患者应仔细随访和监测,不仅要关注甲状腺状态,还要尽早发现该药物潜在的严重并发症。早期诊断并及时停用PTU治疗对于改善预后至关重要。此外,还对PTU诱发的ANCA阳性血管炎的关键方面进行了综述。

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