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进一步描述 ATP6V0A2 相关的常染色体隐性先天性皮肤松弛症。

Further characterization of ATP6V0A2-related autosomal recessive cutis laxa.

机构信息

Institut fuer Medizinische Genetik und Humangenetik, Charité-Universitaetsmedizin Berlin, Berlin, Germany.

出版信息

Hum Genet. 2012 Nov;131(11):1761-73. doi: 10.1007/s00439-012-1197-8. Epub 2012 Jul 8.

Abstract

Autosomal recessive cutis laxa (ARCL) syndromes are phenotypically overlapping, but genetically heterogeneous disorders. Mutations in the ATP6V0A2 gene were found to underlie both, autosomal recessive cutis laxa type 2 (ARCL2), Debré type, and wrinkly skin syndrome (WSS). The ATP6V0A2 gene encodes the a2 subunit of the V-type H(+)-ATPase, playing a role in proton translocation, and possibly also in membrane fusion. Here, we describe a highly variable phenotype in 13 patients with ARCL2, including the oldest affected individual described so far, who showed strikingly progressive dysmorphic features and heterotopic calcifications. In these individuals we identified 17 ATP6V0A2 mutations, 14 of which are novel. Furthermore, we demonstrate a localization of ATP6V0A2 at the Golgi-apparatus and a loss of the mutated ATP6V0A2 protein in patients' dermal fibroblasts. Investigation of brefeldin A-induced Golgi collapse in dermal fibroblasts as well as in HeLa cells deficient for ATP6V0A2 revealed a delay, which was absent in cells deficient for the ARCL-associated proteins GORAB or PYCR1. Furthermore, fibroblasts from patients with ATP6V0A2 mutations displayed elevated TGF-β signalling and increased TGF-β1 levels in the supernatant. Our current findings expand the genetic and phenotypic spectrum and suggest that, besides the known glycosylation defect, alterations in trafficking and signalling processes are potential key events in the pathogenesis of ATP6V0A2-related ARCL.

摘要

常染色体隐性先天性皮肤松弛症(ARCL)综合征在表型上具有重叠性,但在遗传学上具有异质性。已经发现 ATP6V0A2 基因突变是常染色体隐性先天性皮肤松弛症 2 型(ARCL2)、Debré 型和皱纹皮肤综合征(WSS)的致病原因。ATP6V0A2 基因编码 V 型 H(+)-ATP 酶的 a2 亚基,在质子转运中发挥作用,可能在膜融合中也发挥作用。在这里,我们描述了 13 名 ARCL2 患者的高度可变表型,其中包括迄今为止描述的最年长的受影响个体,该个体表现出明显的进行性畸形特征和异位钙化。在这些个体中,我们鉴定了 17 种 ATP6V0A2 突变,其中 14 种是新的。此外,我们证明了 ATP6V0A2 定位于高尔基体,并在患者的真皮成纤维细胞中丢失了突变的 ATP6V0A2 蛋白。在真皮成纤维细胞和缺乏 ATP6V0A2 的 HeLa 细胞中研究布雷菲德菌素 A 诱导的高尔基体塌陷,发现存在延迟,而在缺乏 ARCL 相关蛋白 GORAB 或 PYCR1 的细胞中则不存在。此外,ATP6V0A2 突变患者的成纤维细胞表现出 TGF-β 信号的升高,并且上清液中的 TGF-β1 水平增加。我们目前的发现扩展了遗传和表型谱,并表明除了已知的糖基化缺陷外,运输和信号转导过程的改变可能是 ATP6V0A2 相关 ARCL 发病机制中的潜在关键事件。

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