Molecular mechanisms of opsin transport through cilium in photoreceptor cells

为了确定影响光感受器发育和维持的突变，我们使用转GFP基因的斑马鱼品系进行了早期压力筛选。我们发现了四种影响光感受器纤毛发育的突变。我们发现，这些突变会影响其他感觉神经元的纤毛发生，包括鼻窝的嗅觉细胞和内耳的毛细胞。我们还表明，雄性生殖细胞相关激酶MAK调节视网膜光感受器纤毛长度和亚区划。我们的结果表明，MAK对纤毛蛋白的运输、纤毛长度的调节是必不可少的，并且是光感受器长期生存所必需的。

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.