Center for Biochemistry, Medical Faculty, University of Cologne, Cologne, Germany.
Am J Pathol. 2012 Apr;180(4):1581-92. doi: 10.1016/j.ajpath.2012.01.005. Epub 2012 Feb 9.
Kindlin-1 is an adaptor protein that is expressed by most epithelial cells and has been implicated in integrin bidirectional signaling. Mutations in the gene encoding kindlin-1 are associated with Kindler syndrome, a recessively inherited disorder that is characterized by fragile skin. Functionally, a loss of kindlin-1 impairs the adhesion of basal keratinocytes to the extracellular matrix both in vivo and in vitro. In this study, we show that the phenotype of mutant keratinocytes deficient in kindlin-1 is characterized by the modification of the cortical actin network and increased plasticity of the plasma membrane. At the molecular level, expression of several proteins associated with an epithelial phenotype, such as α6β4 integrin, collagen XVII, E-cadherin, and desmoglein-3, is strongly reduced, whereas, surprisingly, laminin 332 is synthesized in larger amounts than in control keratinocytes. In contrast, mesenchymal markers such as vimentin and fibronectin are increased in keratinocytes lacking kindlin-1. The switch in cell plasticity and protein expression was confirmed by siRNA-mediated down-regulation of kindlin-1 in HaCaT epithelial cells. Furthermore, there was up-regulation of matrix metalloproteinases and pro-inflammatory cytokines in kindlin-1-deficient keratinocytes. These results provide new insights into the pathogenic mechanisms that take place in Kindler syndrome. Moreover, the constellation of molecular defects associated with the loss of kindlin-1 may explain the higher incidence of skin cancer observed in patients affected with this disorder.
Kindlin-1 是一种衔接蛋白,大多数上皮细胞都会表达它,并且已经被牵涉到整合素的双向信号传递中。编码 Kindlin-1 的基因突变与 Kindler 综合征有关,这是一种隐性遗传疾病,其特征是皮肤脆弱。从功能上讲,Kindlin-1 的缺失会损害基底角质形成细胞在体内和体外与细胞外基质的黏附。在这项研究中,我们表明,缺乏 Kindlin-1 的突变角质形成细胞的表型特征是皮质肌动蛋白网络的改变和质膜的可塑性增加。在分子水平上,与上皮表型相关的几种蛋白质的表达,如α6β4 整合素、胶原 XVII、E-钙黏蛋白和桥粒芯糖蛋白 3,强烈减少,而令人惊讶的是,层粘连蛋白 332 的合成量比对照角质形成细胞多。相比之下,缺乏 Kindlin-1 的角质形成细胞中,间充质标志物如波形蛋白和纤维连接蛋白增加。通过 siRNA 介导的 HaCaT 上皮细胞中 Kindlin-1 的下调,证实了细胞可塑性和蛋白表达的转换。此外,在缺乏 Kindlin-1 的角质形成细胞中,基质金属蛋白酶和促炎细胞因子的表达上调。这些结果为 Kindler 综合征中发生的致病机制提供了新的见解。此外,与 Kindlin-1 缺失相关的分子缺陷的组合可能解释了在患有这种疾病的患者中观察到的更高的皮肤癌发病率。