Early defects in photoreceptor outer segment morphogenesis in zebrafish ift57, ift88 and ift172 Intraflagellar Transport mutants.

Early defects in photoreceptor outer segment morphogenesis in zebrafish ift57, ift88 and ift172 Intraflagellar Transport mutants.
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DOI:
10.1016/j.visres.2008.12.009
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发表时间:
2009-02
期刊:
影响因子:
1.8
通讯作者:
Perkins, Brian D.
Perkins, Brian D.
中科院分区:
心理学3区
文献类型:
--
作者:
Sukumaran, Sujita;Perkins, Brian D.

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鞭毛内转运(IFT)是指发生在真核纤毛结构中的高度保守的过程。在脊椎动物光感受器中,IFT介导蛋白质运输到外节。IFT颗粒是一种多亚基复合物,许多单个组分的突变会导致感光细胞缺陷。在斑马鱼中,ift57、ift88和ift172基因的突变导致受精后5天(dpf)视网膜变性。尽管这些突变对光感受器存活的影响已经被描述,但早期发育形态发生仍然知之甚少。我们用透射电子显微镜和免疫组化检查这些突变体在60,72和96小时后受精(hpf),并描述早期感光细胞形态发生缺陷。
Intraflagellar Transport (IFT) refers to a highly conserved process occurring in eukaryotic ciliated structures. In vertebrate photoreceptors, IFT mediates protein trafficking to the outer segments. The IFT particle is a multi-subunit complex and mutations in many individual components causes photoreceptor defects. In zebrafish, mutations in the ift57, ift88 and ift172 genes result in retinal degeneration by 5 days post fertilization (dpf). Although the effects of these mutations on photoreceptor survival have been described, early developmental morphogenesis remains poorly understood. We used transmission electron microscopy and immunohistochemistry to examine these mutants at 60, 72, and 96 hours post fertilization (hpf) and describe early photoreceptor morphogenesis defects.
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