Syntrophin-2 is required for eye development in Drosophila
Syntrophin-2 is required for eye development in Drosophila
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DOI：
10.1016/j.yexcr.2009.10.009
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发表时间：
2010-01-15
影响因子：
3.7
通讯作者：
Yamaguchi, Masamitsu
中科院分区：
文献类型：
--
作者：
Nagai, Rika;Hashimoto, Reina;Yamaguchi, Masamitsu
肌营养蛋白是肌营养不良蛋白糖蛋白复合物（DGC）的组分，其由肌营养不良症的致病基因编码。DGC被认为不仅在将肌动蛋白细胞骨架连接到细胞外基质，为细胞膜提供稳定性方面发挥作用，而且在信号转导中发挥作用。由于它们与各种不同的分子结合，已经表明突触营养蛋白是将信号蛋白募集到膜和DGC的衔接蛋白。然而，在体内的关键作用仍然难以捉摸。果蝇Syntrophin-2（Syn 2）是人β 1/γ 2-syntrophins的直向同源物。Western免疫印迹分析显示Syn 2在整个发育过程中都有表达，在成人头部的表达水平特别高。在Syn 2基因敲除的成年果蝇中观察到形态学畸变，视网膜缺乏伸长和横纹肌畸形。此外，Syn 2基因敲除苍蝇表现出过度凋亡的三龄幼虫和蛹视网膜中的肌动蛋白定位的改变。遗传杂交与果蝇缺乏症股票的集合，使我们能够确定7个基因组区域，其中删除引起增强的坚韧的眼睛表型诱导Syn 2敲低。这些信息将有助于识别果蝇中的Syn 2调节因子，并阐明Syn 2在眼睛发育中的作用。(C)2009 Elsevier Inc. All rights reserved.
Syntrophins are components of the dystrophin glycoprotein complex (DGC), which is encoded by causative genes of muscular dystrophies. The DGC is thought to play roles not only in linking the actin cytoskeleton to the extracellular matrix, providing stability to the cell membrane, but also in signal transduction. Because of their binding to a variety of different molecules, it has been suggested that syntrophins are adaptor proteins recruiting signaling proteins to membranes and the DGC. However, critical roles in vivo remain elusive. Drosophila Syntrophin-2 (Syn2) is an orthologue of human -gamma 1/gamma 2-syntrophins. Western immunoblot analysis here showed Syn2 to be expressed throughout development, with especially high levels in the adult head. Morphological aberrations were observed in Syn2 knockdown adult flies, with lack of retinal elongation and malformation of rhabdomeres. Furthermore, Syn2 knockdown flies exhibited excessive apoptosis in third instar larvae and alterations in the actin localization in the pupal retinae. Genetic crosses with a collection of Drosophila deficiency stocks allowed us to identify seven genomic regions, deletions of which caused enhancement of the Tough eye phenotype induced by Syn2 knockdown. This information should facilitate identification of Syn2 regulators in Drosophila and clarification of roles of Syn2 in eye development. (C) 2009 Elsevier Inc. All rights reserved.