Department of Biological Sciences, University of Notre Dame, Notre Dame, Indiana 46556, USA.
J Biol Chem. 2013 Jan 11;288(2):886-93. doi: 10.1074/jbc.M112.417089. Epub 2012 Nov 19.
We previously isolated a dominant mutation, night blindness b (nbb), which causes a late onset of retinal dopaminergic cell degeneration in zebrafish. In this study, we cloned the zebrafish nbb locus. Sequencing results revealed that nbb is a homolog of the vertebrate SCL/TAL1 interrupting locus (Stil). The Stil gene has been shown to play important roles in the regulation of vertebrate embryonic neural development and human cancer cell proliferation. In this study, we demonstrate that functional expression of Stil is also required for neural survival. In zebrafish, decreased expression of Stil resulted in increased toxic susceptibility of retinal dopaminergic cells to 6-hydroxydopamine. Increases in Stil-mediated Shh signaling transduction (i.e. by knocking down the Shh repressor Sufu) prevented dopaminergic cell death induced by neurotoxic insult. The data suggest that the oncogene Stil also plays important roles in neural protection.
我们之前分离出一个显性突变体,夜盲症 b(nbb),它导致斑马鱼视网膜多巴胺能细胞退化的迟发。在这项研究中,我们克隆了斑马鱼 nbb 基因座。测序结果表明,nbb 是脊椎动物 SCL/TAL1 中断基因座(Stil)的同源物。Stil 基因已被证明在调节脊椎动物胚胎神经发育和人类癌细胞增殖中发挥重要作用。在这项研究中,我们证明 Stil 的功能表达对于神经存活也是必需的。在斑马鱼中,Stil 表达的减少导致视网膜多巴胺能细胞对 6-羟多巴胺的毒性易感性增加。增加 Stil 介导的 Shh 信号转导(例如通过敲低 Shh 抑制剂 Sufu)可防止神经毒性损伤诱导的多巴胺能细胞死亡。数据表明,致癌基因 Stil 也在神经保护中发挥重要作用。