Kashyap Subhash, Shanker Vinay, Sharma Neelam
Department of Dermatology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India.
Department of Pathology, Indira Gandhi Medical College, Shimla, Himachal Pradesh, India.
Indian J Dermatol. 2015 Jul-Aug;60(4):421. doi: 10.4103/0019-5154.160525.
Ectodermal dysplasia/skin fragility syndrome (ED-SFS) is a newly described autosomal recessive disorder characterized by skin fragility and blistering, palmoplantar keratoderma, abnormal hair growth, nail dystrophy, and occasionally defective sweating. It results from mutations in the PKP1 gene encoding plakophilin 1 (PKP1), which is an important component of stratifying epithelial desmosomes and a nuclear component of many cell types. Only 12 cases of this rare genodermatosis have been reported so far. We present an unusual case of ED-SFS in a 12-year boy who was normal at birth but subsequently developed skin fragility, hair and nail deformities, abnormal dentition, palmoplantar keratoderma, and abnormal sweating but no systemic abnormality.
外胚层发育不良/皮肤脆性综合征(ED-SFS)是一种新描述的常染色体隐性疾病,其特征为皮肤脆弱、水疱形成、掌跖角化病、毛发异常生长、指甲营养不良,偶尔还有出汗缺陷。它是由编码桥粒芯蛋白1(PKP1)的PKP1基因突变引起的,PKP1是分层上皮桥粒的重要组成部分,也是许多细胞类型的核成分。迄今为止,仅报道了12例这种罕见的遗传性皮肤病。我们报告了一例12岁男孩的罕见ED-SFS病例,该男孩出生时正常,但随后出现皮肤脆弱、毛发和指甲畸形、牙齿异常、掌跖角化病和出汗异常,但无全身异常。