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匐行性穿通性弹力纤维病与威尔逊病:青霉胺长期治疗的一种罕见但可预测的后果

[Elastosis Perforans Serpiginosa and Wilson Disease: A Rare but Predictable Consequence of Long-term Therapy with D-Penicillamine].

作者信息

Castro Pinho André, Cardoso José Carlos, Gouveia Miguel, Oliveira Hugo

机构信息

Serviço de Dermatologia. Hospitais da Universidade de Coimbra. Centro Hospitalar e Universitário de Coimbra. Coimbra.

出版信息

Acta Med Port. 2016 Mar;29(3):227-230. doi: 10.20344/amp.6749. Epub 2016 Mar 31.

DOI:10.20344/amp.6749
PMID:27285100
Abstract

Elastosis perfurans serpiginosa is a rare perforating dermatosis found primarily in adolescents and young adults, characterized by transepidermal elimination of abnormal elastic fibers. The only drug known capable of inducing elastosis perfurans serpiginosa is D-penicillamine. We report the case of a 52 year-old woman with keratotic papules arranged in an annular pattern with central clearing and centrifugal growth, located in the anterior cervical region. The patient was chronically treated with D-penicillamine for Wilson disease. Lesion biopsy showed transepidermal elimination of thickened, eosinophilic, branched, sawtooth-like elastic fibers. The clinical and pathological findings were consistent with elastosis perfurans serpiginosa secondary to D-penicillamine. It is estimated that elastosis perfurans serpiginosa occurs in 1% of patients treated with D-penicillamine. By blocking directly or indirectly the desmosine cross-links between elastin molecules, D-penicillamine leads to the synthesis of abnormal dermal and extracutaneous elastic fibers. Elastosis perfurans serpiginosa may be the first manifestation of a multisystemic degenerative process of elastic connective tissue.

摘要

匐行性穿通性弹力纤维病是一种罕见的穿通性皮肤病,主要见于青少年和青年,其特征为异常弹性纤维经表皮排出。已知唯一能诱发匐行性穿通性弹力纤维病的药物是D-青霉胺。我们报告一例52岁女性病例,其颈部前方区域有角化性丘疹,呈环状排列,中央有消退区并呈离心性生长。该患者因威尔逊病长期接受D-青霉胺治疗。病变活检显示增厚的、嗜酸性的、分支状的、锯齿状弹性纤维经表皮排出。临床和病理表现符合D-青霉胺继发的匐行性穿通性弹力纤维病。据估计,在接受D-青霉胺治疗的患者中,匐行性穿通性弹力纤维病的发生率为1%。D-青霉胺通过直接或间接阻断弹性蛋白分子之间的锁链素交联,导致异常的真皮和皮肤外弹性纤维合成。匐行性穿通性弹力纤维病可能是弹性结缔组织多系统退行性过程的首发表现。

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Acta Med Port. 2016 Mar;29(3):227-230. doi: 10.20344/amp.6749. Epub 2016 Mar 31.
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