Molecular mechanisms of opsin transport through cilium in photoreceptor cells

为了鉴定影响光感受器发育和维持的突变，我们使用GFP转基因斑马鱼系进行了早期压力筛选。我们发现了四种影响感光细胞纤毛发育的突变。我们发现，这些突变影响其他感觉神经元的纤毛发生，包括鼻窝中的嗅觉细胞和内耳中的毛细胞。我们还发现，男性生殖细胞相关激酶，Mak，调节视网膜感光细胞纤毛长度和亚区室化。我们的研究结果表明，Mak是必不可少的纤毛蛋白运输，纤毛长度的调节，是所需的长期生存的光感受器。

To identify mutations that affect the photoreceptor development and maintenance, we performed early pressure screen using a GFP transgenic line of zebrafish. We showed four mutations which affect ciliary development in photoreceptors. We found that these mutations affect in the ciliogenesis of other sensory neurons including olfactory cells in the nasal pit and hair cells in the inner ear. We also showed that male germ cell-associated kinase, Mak, regulates retinal photoreceptor ciliary length and subcompartmentalization. Our results suggest that Mak is essential for ciliary protein transport, regulation of ciliary length, and is required for the long-term survival of photoreceptors.