Radial spoke proteins regulate otolith formation during early zebrafish development
Radial spoke proteins regulate otolith formation during early zebrafish development
复制标题
径向辐条蛋白在斑马鱼早期发育过程中调节耳石形成
DOI：
10.1096/fj.201701359r
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发表时间：
2018-07-01
期刊：
影响因子：
4.8
通讯作者：
Zhao, Chengtian
中科院分区：
文献类型：
--
作者：
Han, Xiao;Xie, Haibo;Zhao, Chengtian
放射辐条在结构上是保守的大分子复合物，对9 + 2运动纤毛的运动至关重要。在衣藻属物种中，放射辐条蛋白的突变会导致纤毛运动缺陷。然而，放射辐条蛋白在胚胎发育过程中的功能却鲜为人知。在此，我们研究了一种新型放射辐条蛋白——富含亮氨酸重复序列蛋白23（Lrrc23）在斑马鱼胚胎发育过程中的作用。lrrc23的突变导致在早期耳部发育过程中出现选择性耳石形成缺陷。在放射辐条头部3同源物（rsph3）和放射辐条头部4同源物A（rsph4a）放射辐条突变体中也存在类似的耳石缺陷。值得注意的是，放射辐条蛋白突变特异性地影响了耳泡（OV）中的纤毛运动，而其他器官中的运动纤毛功能正常。通过高速视频显微镜，我们发现耳泡中运动纤毛的形成是随机且短暂的。重要的是，耳泡中的所有运动纤毛都呈环形摆动，这与典型的9 + 2运动纤毛的平面摆动模式不同。我们确定了耳泡发育过程中运动纤毛形成的关键时间框架。最后，我们表明放射辐条蛋白的功能在斑马鱼和四膜虫之间是保守的。总之，我们的研究结果表明，放射辐条蛋白对耳泡中的纤毛运动至关重要，并且放射辐条调节的耳泡运动纤毛在斑马鱼早期胚胎发育过程中代表了一种独特类型的纤毛。韩，X.；谢，H.；王，Y.；赵，C. 放射辐条蛋白在斑马鱼早期发育过程中调节耳石形成。
Radial spokes are structurally conserved, macromolecular complexes that are essential for the motility of 9 + 2 motile cilia. In Chlamydomonas species, mutations in radial spoke proteins result in ciliary motility defects. However, little is known about the function of radial spoke proteins during embryonic development. Here, we investigated the role of a novel radial spoke protein, leucine‐rich repeat containing protein 23 (Lrrc23), during zebrafish embryonic development. Mutations in lrrc23 resulted in a selective otolith formation defect during early ear development. Similar otolith defects were also present in the radial spoke head 3 homolog (rsph3)and radial spoke head 4 homolog A (rsph4a) radial spoke mutants. Notably, the radial spoke protein mutations specifically affected ciliary motility in the otic vesicle (OV), whereas motile cilia in other organs functioned normally. Via highspeed video microscopy, we found that motile cilia formation was stochastic and transient in the OV. Importantly, all the motile cilia in the OV beat circularly, in contrast to the planar beating pattern of typical 9 + 2 motile cilia. We identified the key time frame for motile cilia formation during OV development. Finally, we showed that the functions of radial spoke proteins were conserved between zebrafish and Tetrahymena. Together, our results suggest that radial spoke proteins are essential for ciliary motility in the OV and that radial spoke‐regulated OV motile cilia represent a unique type of cilia during early zebrafish embryonic development.—Han, X., Xie, H., Wang, Y., Zhao, C. Radial spoke proteins regulate otolith formation during early zebrafish development. FASEB J. 32, 3984–3992 (2018). www.fasebj.org