Discovery of a Vertebrate-Specific Factor that Processes Flagellar Glycolytic Enolase during Motile Ciliogenesis
Discovery of a Vertebrate-Specific Factor that Processes Flagellar Glycolytic Enolase during Motile Ciliogenesis
复制标题
发现在运动纤毛发生过程中处理鞭毛糖酵解烯醇酶的脊椎动物特异性因子
DOI：
10.1016/j.isci.2020.100992
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发表时间：
2020
期刊：
影响因子：
5.8
通讯作者：
Takeda Sen
中科院分区：
文献类型：
--
作者：
Narita Keishi;Nagatomo Hiroaki;Kozuka-Hata Hiroko;Oyama Masaaki;Takeda Sen
活动的纤毛和鞭毛的形成和功能需要三磷酸腺苷。虽然糖酵解酶是鞭毛蛋白质组的组成部分，但它们是如何转运到鞭毛的尚不清楚。在这里，我们发现了仅在脊椎动物中发现的功能未注释的基因4833427G06Rik(C11orf88)的表达模式，该基因在这里被命名为Hoatzin(Hoatz)，表明其产物与活动的纤毛和鞭毛具有功能性关联。HoatzKackout(KO)小鼠以常染色体隐性方式发生脑积水和男性不育，室管膜纤毛经常表现为无序的轴突，在细胞分化过程中减少与精子细胞鞭毛塌陷相关的运动能力。HOATZ与某些蛋白质有关，包括鞭毛糖酵解酶ENO4。在HoatzKO小鼠的睾丸中，未成熟的ENO4聚集在发育中的精子细胞的异常细胞质斑点中。这些数据表明，HOATZ是脊椎动物活动的纤毛发生和鞭毛发生所必需的，它通过介导ENO4的成熟来实现。
Motile cilia and flagella require ATP for their formation and function. Although glycolytic enzymes are components of flagellar proteomes, how they translocate to flagella is unknown. Here we show that the expression pattern of the functionally nonannotated gene4833427G06Rik(C11orf88), which is found only in vertebrates and is designated here as Hoatzin (Hoatz), suggests a functional association of its product with motile cilia and flagella.Hoatzknockout (KO) mice developed hydrocephalus and male infertility in an autosomal recessive manner, and the ependymal cilia frequently showed disorganized axonemes, reducing motility associated with collapsed spermatid flagella during cytodifferentiation. HOATZ was associated with certain proteins, including the flagellar glycolytic enzyme ENO4. In the testes of theHoatzKO mice, the immature form of ENO4 accumulated in abnormal cytoplasmic puncta of developing spermatids. These data indicate that HOATZ is required for motile ciliogenesis and flagellar genesis in vertebrates by mediating the maturation of ENO4.