Wolff Christian, Roy Sudipto, Lewis Katharine E, Schauerte Heike, Joerg-Rauch Gerd, Kirn Annette, Weiler Christian, Geisler Robert, Haffter Pascal, Ingham Philip W
Center for Developmental Genetics, School of Medicine and Biomedical Sciences, University of Sheffield, Western Bank, Sheffield S10 2TN, UK.
Genes Dev. 2004 Jul 1;18(13):1565-76. doi: 10.1101/gad.296004. Epub 2004 Jun 15.
Signaling by lipid-modified secreted glycoproteins of the Hedgehog family play fundamental roles during pattern formation in animal development and in humans; dysfunction of Hedgehog pathway components is frequently associated with a variety of congenital abnormalities and cancer. Transcriptional regulation of Hedgehog target genes is mediated by members of the Gli zinc-finger transcription factors. The relative nuclear concentrations of Gli activator (Gli(act)) and repressor (Gli(rep)) forms, together with their nucleocytoplasmic trafficking, appear to be critical determinants for target gene expression. Whereas such stringent controls of Gli activity are critical in ensuring appropriate levels of pathway activation, the mechanisms by which these processes are regulated remain inadequately understood. Here, using genetic analysis, we show that the zebrafish iguana gene product acts downstream of the Smoothened protein to modulate Gli activity in the somites of the developing embryo. Positional cloning reveals that iguana encodes the zebrafish ortholog of Dzip1, a novel zinc-finger/coiled-coil domain protein that we show can shuttle between the cytoplasm and nucleus in a manner correlated with Hedgehog pathway activity.
刺猬索尼克家族的脂质修饰分泌糖蛋白信号传导在动物发育和人类的模式形成过程中发挥着重要作用;刺猬索尼克信号通路成分的功能障碍常常与多种先天性异常和癌症相关。刺猬索尼克靶基因的转录调控由Gli锌指转录因子家族成员介导。Gli激活剂(Gli(act))和抑制剂(Gli(rep))形式的相对核浓度,以及它们在核质间的运输,似乎是靶基因表达的关键决定因素。虽然对Gli活性的这种严格控制对于确保信号通路激活的适当水平至关重要,但这些过程的调控机制仍未得到充分理解。在这里,我们通过遗传分析表明,斑马鱼鬣蜥基因产物在平滑蛋白下游发挥作用,调节发育中胚胎体节内的Gli活性。定位克隆显示,鬣蜥基因编码Dzip1的斑马鱼直系同源物,Dzip1是一种新型锌指/卷曲螺旋结构域蛋白,我们发现它能够以与刺猬索尼克信号通路活性相关的方式在细胞质和细胞核之间穿梭。