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首页> 外文期刊>PLoS Genetics >Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice
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Nexin-Dynein regulatory complex component DRC7 but not FBXL13 is required for sperm flagellum formation and male fertility in mice

机译：Nexin-Dynein调节络合物组分DRC7但不是FBXL13需要小鼠的精子鞭毛形成和雄性生育

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Flagella and cilia are evolutionarily conserved cellular organelles. Abnormal formation or motility of these organelles in humans causes several syndromic diseases termed ciliopathies. The central component of flagella and cilia is the axoneme that is composed of the ‘9+2’ microtubule arrangement, dynein arms, radial spokes, and the Nexin-Dynein Regulatory Complex (N-DRC). The N-DRC is localized between doublet microtubules and has been extensively studied in the unicellular flagellate Chlamydomonas . Recently, it has been reported that TCTE1 (DRC5), a component of the N-DRC, is essential for proper sperm motility and male fertility in mice. Further, TCTE1 has been shown to interact with FBXL13 (DRC6) and DRC7; however, functional roles of FBXL13 and DRC7 in mammals have not been elucidated. Here we show that Fbxl13 and Drc7 expression are testes-enriched in mice. Although Fbxl13 knockout (KO) mice did not show any obvious phenotypes, Drc7 KO male mice were infertile due to their short immotile spermatozoa. In Drc7 KO spermatids, the axoneme is disorganized and the ‘9+2’ microtubule arrangement was difficult to detect. Further, other N-DRC components fail to incorporate into the flagellum without DRC7. These results indicate that Drc7 , but not Fbxl13 , is essential for the correct assembly of the N-DRC and flagella.
机译：鞭毛和纤毛是进化的保守细胞细胞器。这些细胞器在人体中的形成或动力导致若干综合征疾病称为纤毛病。鞭毛和纤毛的中央分量是由'9 + 2'微管布置，Dynein臂，径向辐条和Nexin-Dynein调节综合体（N-DRC）组成的轴突。 N-DRC位于双峰微管之间，并在单细胞鞭素衣原体中进行了广泛研究。最近，据报道，TCTE1（DRC5）是N-DRC的组分对于小鼠的适当精子运动和男性生育是至关重要的。此外，已显示TCTE1与FBXL13（DRC6）和DRC7相互作用;然而，FBXL13和DRC7在哺乳动物中的功能作用尚未阐明。在这里，我们表明FBXL13和DRC7表达是富集的小鼠睾丸。虽然FBXL13敲除（KO）小鼠没有显示出任何明显的表型，但由于其短的Immotile精子，DRC7 KO雄性小鼠的不含育种。在DRC7 KO精子中，轴突被混乱，难以检测'9 + 2'微管布置。此外，其他N-DRC组件未结合到没有DRC7的鞭毛中。这些结果表明DRC7但不是FBXL13，对于N-DRC和鞭毛的正确组装至关重要。

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