Chao Sheau-Chiou, Yang Mei-Hui, Lee Shu-Fen
Department of Dermatology, National Cheng-Kung University Hospital, 138 Sheng-Li Road, Tainan, Taiwan.
J Formos Med Assoc. 2002 Apr;101(4):287-90.
Epidermolysis bullosa simplex (EBS) is a group of hereditary bullous diseases characterized by intraepidermal blistering due to mechanical stress-induced degeneration of basal keratinocytes. Three major subtypes have been identified with autosomal dominant inheritance: the Weber-Cockayne type, the Köbner type (EBS-K), and the Dowling-Meara type. All three EBS subtypes are caused by mutations in either keratin 5 or keratin 14, the major keratins expressed in the basal layer of the epidermis. We describe a 25-year-old male with easy blistering after trauma over the whole body from the age of 4 to 5 years. According to the clinicopathologic findings, EBS-K was diagnosed. Mutational analysis revealed a novel keratin 14 mutation (1237G-->A) that produces a conservative amino acid change (alanine to threonine) at position 413 (A413T) of the 2B helix.
单纯性大疱性表皮松解症(EBS)是一组遗传性大疱性疾病,其特征是由于机械应力诱导基底角质形成细胞变性而导致表皮内水疱形成。已确定三种主要亚型呈常染色体显性遗传:Weber-Cockayne型、Köbner型(EBS-K)和Dowling-Meara型。所有这三种EBS亚型均由角蛋白5或角蛋白14的突变引起,这两种是表皮基底层中表达的主要角蛋白。我们描述了一名25岁男性,从4至5岁起全身外伤后容易出现水疱。根据临床病理结果,诊断为EBS-K。突变分析发现了一种新的角蛋白14突变(1237G→A),该突变在2B螺旋的第413位(A413T)产生保守的氨基酸变化(丙氨酸变为苏氨酸)。