INSERM UMR 1163, Laboratory of Genetic Skin Diseases, Imagine Institute for Genetic Diseases, Paris, France; University Paris Descartes, Sorbonne Paris Cité, Paris, France.
INSERM UMR 1163, Laboratory of Genetic Skin Diseases, Imagine Institute for Genetic Diseases, Paris, France; University Paris Descartes, Sorbonne Paris Cité, Paris, France; Department of Genetics, Necker Enfants Malades Hospital, Paris, France.
J Invest Dermatol. 2015 Jun;135(6):1475-1478. doi: 10.1038/jid.2014.535.
Erythrokeratodermia variabilis et progressiva (EKVP) is a genodermatosis with clinical and genetic heterogeneity, most often transmitted in an autosomal dominant manner, caused by mutations in GJB3 and GJB4 genes encoding connexins (Cx)31 and 30.3, respectively. In this issue, Boyden et al. (2015) report for the first time de novo dominant mutations in GJA1 encoding the ubiquitous Cx43 in patients with EKVP. These results expand the genetic heterogeneity of EKVP and the human disease phenotypes associated with GJA1 mutations. They disclose that EKVP is allelic to oculo-dento-digital dysplasia, a rare syndrome previously known to be caused by dominant GJA1 mutations.
进行性全身性红斑角化症(Erythrokeratodermia variabilis et progressiva,EKVP)是一种具有临床和遗传异质性的遗传性皮肤病,最常以常染色体显性方式遗传,由编码连接蛋白(Connexin,Cx)31 和 Cx30.3 的 GJB3 和 GJB4 基因突变引起。在本期中,Boyden 等人(2015)首次报道了 EKVP 患者中普遍存在的 GJA1 编码连接蛋白(Connexin,Cx)43 的从头显性突变。这些结果扩展了 EKVP 的遗传异质性以及与 GJA1 突变相关的人类疾病表型。它们揭示 EKVP 与眼-牙-指(趾)发育不良是等位基因相关的,后者是一种先前已知由显性 GJA1 突变引起的罕见综合征。