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成人获得性大疱性表皮松解症(EB)伴先前未识别的轻度营养不良性 EB 和 COL7A1 双等位基因突变

Epidermolysis Bullosa (EB) Acquisita in an Adult Patient with Previously Unrecognized Mild Dystrophic EB and Biallelic COL7A1 Mutations.

机构信息

Laboratory of Molecular and Cell Biology, Instituto Dermopatico dell'Immacolata-IRCCS, Via dei Monti di Creta, 104, IT-00167 Rome, Italy.

出版信息

Acta Derm Venereol. 2018 Apr 16;98(4):411-415. doi: 10.2340/00015555-2851.

DOI:10.2340/00015555-2851
PMID:29182795
Abstract

Circulating anti-type VII collagen autoantibodies are frequently detected in patients with recessive dystrophic epidermolysis bullosa (RDEB). However, evidence supporting their pathogenic role in inducing epidermolysis bullosa acquisita (EBA) has been provided for only one individual with dominant dystrophic epidermolysis bullosa (DDEB). We describe here a patient who presented with dystrophic toenails since early childhood and developed trauma-induced skin blisters and oral erosions at age 26 years. Direct immunofluorescence showed IgG deposits with a u-serrated pattern along the cutaneous basement membrane zone, while no change in the expression of collagen VII could be detected by antigen mapping. High-titre anti-collagen VII antibodies were detected by enzyme-linked immunoassay (ELISA). In parallel, sequencing of epidermolysis bullosa (EB) genes identified compound heterozygous COL7A1 missense c.410G>A (p.Arg137Gln) and splicing c.3674C>T (p.Ala1225_Gln1241del) mutations, previously unrecognized in dystrophic epidermolysis bullosa (DEB). Thus, our patient had RDEB "nails-only" and developed mechanobullous EBA in adulthood. These data support a pathogenic role of circulating autoantibodies to collagen VII in inducing EBA in selected patients with DEB. Unforeseen worsening of skin symptoms in DEB should prompt laboratory investigations for EBA.

摘要

循环抗 VII 型胶原自身抗体在隐性营养不良型大疱性表皮松解症(RDEB)患者中经常被检测到。然而,仅有一例显性营养不良型大疱性表皮松解症(DDEB)患者的证据支持其在诱导获得性大疱性表皮松解症(EBA)中的致病作用。我们在此描述了一位患者,他从小就患有营养不良型脚趾甲,并在 26 岁时因创伤导致皮肤水疱和口腔糜烂。直接免疫荧光显示 IgG 沉积物呈 u 形锯齿状模式沿着皮肤基底膜带,而通过抗原定位未检测到 VII 型胶原表达的变化。通过酶联免疫吸附试验(ELISA)检测到高滴度的抗 VII 型胶原抗体。同时,大疱性表皮松解症(EB)基因的测序发现 COL7A1 复合杂合错义 c.410G>A(p.Arg137Gln)和剪接 c.3674C>T(p.Ala1225_Gln1241del)突变,以前在营养不良型大疱性表皮松解症(DEB)中未被识别。因此,我们的患者患有 RDEB“仅指甲”,并在成年后发展为机械性大疱性 EBA。这些数据支持循环抗 VII 型胶原自身抗体在诱导特定 DEB 患者 EBA 中的致病作用。DEB 患者皮肤症状的意外恶化应促使进行 EBA 的实验室检查。

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