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首页> 外文期刊>Molecular and Cellular Biology >Mice Deficient in the Axonemal Protein Tektin-t Exhibit Male Infertility and Immotile-Cilium Syndrome Due to Impaired Inner Arm Dynein Function
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Mice Deficient in the Axonemal Protein Tektin-t Exhibit Male Infertility and Immotile-Cilium Syndrome Due to Impaired Inner Arm Dynein Function

机译：缺乏轴索蛋白Tektin-t的小鼠表现出由于臂内动力蛋白功能受损而引起的男性不育和运动性Cilium综合征。

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摘要

The haploid germ cell-specific Tektin-t protein is a member of the Tektin family of proteins that form filaments in flagellar, ciliary, and axonemal microtubules. To investigate the physiological role of Tektin-t, we generated mice with a mutation in the tektin-t gene. The homozygous mutant males were infertile, while the females were fully fertile. Sperm morphology and function were abnormal, with frequent bending of the sperm flagella and marked defects in motility. In vitro fertilization assays showed that the defective spermatozoa were able to fertilize eggs. Electron microscopic examination showed that the dynein inner arm structure was disrupted in the sperm flagella of tektin-t-deficient mice. Furthermore, homozygous mutant mice had functionally defective tracheal cilia, as evidenced by altered dynein arm morphology. These results indicate that Tektin-t participates in dynein inner arm formation or attachment and that the loss of Tektin-t results in impaired motility of both flagella and cilia. Therefore, the tektin-t gene is one of the causal genes for immotile-cilium syndrome/primary ciliary dyskinesia.
机译：单倍体生殖细胞特异性Tektin-t蛋白是Tektin蛋白家族的成员，该蛋白在鞭毛，睫状和轴突微管中形成细丝。为了研究Tektin-t的生理作用，我们生成了在 tektin-t 基因中具有突变的小鼠。纯合突变的雄性不育，而雌性则完全受精。精子形态和功能异常，精子鞭毛频繁弯曲，运动能力明显受损。体外受精测定表明，有缺陷的精子能够使卵受精。电子显微镜检查显示，在缺乏 tektin-t-缺陷小鼠的精子鞭毛中，动力蛋白的内臂结构被破坏。此外，纯合突变小鼠具有功能缺陷的气管纤毛，如动力蛋白形态改变所证明。这些结果表明Tektin-t参与了动力蛋白内臂的形成或附着，并且Tektin-t的丧失导致鞭毛和纤毛的运动能力受损。因此， tektin-t 基因是免疫性纤毛综合症/原发性睫状运动障碍的致病基因之一。

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