Intraflagellar Transport Proteins Are Essential for Cilia Formation and for Planar Cell Polarity

Intraflagellar Transport Proteins Are Essential for Cilia Formation and for Planar Cell Polarity
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DOI:
10.1681/asn.2009091001
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发表时间:
2010-08-01
影响因子:
13.6
通讯作者:
Sun, Zhaoxia
Sun, Zhaoxia
中科院分区:
医学1区
文献类型:
--
作者:
Cao, Ying;Park, Alice;Sun, Zhaoxia

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高度保守的鞭毛内转运(IFT)蛋白对于多种生物的纤毛形成是必不可少的,但令人惊讶的是,在多个斑马鱼ift突变体中却形成了纤毛。在这里,我们检测到斑马鱼体内IFT基因产物的母体沉积,并发现纤毛组装只发生在早期发育阶段,这支持了母体贡献的IFT基因产物掩盖了IFT蛋白在初始发育阶段的功能的观点。此外,ift突变体的原肾管多纤毛细胞中的基底层排列紊乱,提示存在平面细胞极性缺陷(PCP)。PCP的核心成分pk1的缺失同样会导致肾囊肿的形成和基底体的破坏。此外,我们还发现多个ift基因与pk1存在基因上的相互作用。综上所述,这些数据表明IFT蛋白在斑马鱼的纤毛形成和平面细胞极性中起着保守的作用。
The highly conserved intraflagellar transport (IFT) proteins are essential for cilia formation in multiple organisms, but surprisingly, cilia form in multiple zebrafish ift mutants. Here, we detected maternal deposition of ift gene products in zebrafish and found that ciliary assembly occurs only during early developmental stages, supporting the idea that maternal contribution of ift gene products masks the function of IFT proteins during initial development. In addition, the basal bodies in multiciliated cells of the pronephric duct in ift mutants were disorganized, with a pattern suggestive of defective planar cell polarity (PCP). Depletion of pk1, a core PCP component, similarly led to kidney cyst formation and basal body disorganization. Furthermore, we found that multiple ift genes genetically interact with pk1. Taken together, these data suggest that IFT proteins play a conserved role in cilia formation and planar cell polarity in zebrafish.