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首页> 外文期刊>Biology Open >The Golgi matrix protein giantin is required for normal cilia function in zebrafish
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The Golgi matrix protein giantin is required for normal cilia function in zebrafish

机译：高尔基体蛋白巨蛋白是斑马鱼正常纤毛功能所必需的

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The Golgi is essential for glycosylation of newly synthesised proteins including almost all cell-surface and extracellular matrix proteoglycans. Giantin, encoded by the?golgb1?gene, is a member of the golgin family of proteins that reside within the Golgi stack, but its function remains elusive. Loss of function of giantin in rats causes osteochondrodysplasia; knockout mice show milder defects, notably a cleft palate.?In vitro, giantin has been implicated in Golgi organisation, biosynthetic trafficking, and ciliogenesis. Here we show that loss of function of giantin in zebrafish, using either morpholino or knockout techniques, causes defects in cilia function. Giantin morphants have fewer cilia in the neural tube and those remaining are longer. Mutants have the same number of cilia in the neural tube but these cilia are also elongated. Scanning electron microscopy shows that loss of giantin results in an accumulation of material at the ciliary tip, consistent with a loss of function of retrograde intraflagellar transport. Mutants show milder defects than morphants consistent with adaptation to loss of giantin.
机译：高尔基体对于新合成的蛋白质（包括几乎所有细胞表面和细胞外基质蛋白聚糖）的糖基化至关重要。由“ golgb1”基因编码的巨蛋白是高尔基体中的高尔金蛋白家族的成员，但其功能仍然难以捉摸。大鼠海洛因功能丧失导致骨软骨发育不良;敲除小鼠显示出较轻的缺陷，特别是a裂。在体外，巨蛋白与高尔基体的组织，生物合成的运输和纤毛发生有关。在这里我们表明，使用吗啉代或敲除技术在斑马鱼中巨蛋白功能的丧失会导致纤毛功能的缺陷。巨人变体在神经管中的纤毛较少，而剩余的则更长。突变体在神经管中具有相同数量的纤毛，但这些纤毛也被拉长。扫描电子显微镜显示巨蛋白的损失导致睫状体尖端物质的积累，这与逆行鞭毛内运输功能的丧失相一致。突变体显示出比变异体更弱的缺陷，这与适应巨蛋白的损失相一致。

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