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Zebrafish: a vertebrate tool for studying basal body biogenesis, structure, and function

机译：斑马鱼：研究基础身体生物发生，结构和功能的脊椎动物工具

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Understanding the role of basal bodies (BBs) during development and disease has been largely overshadowed by research into the function of the cilium. Although these two organelles are closely associated, they have specific roles to complete for successful cellular development. Appropriate development and function of the BB are fundamental for cilia function. Indeed, there are a growing number of human genetic diseases affecting ciliary development, known collectively as the ciliopathies. Accumulating evidence suggests that BBs establish cell polarity, direct ciliogenesis, and provide docking sites for proteins required within the ciliary axoneme. Major contributions to our knowledge of BB structure and function have been provided by studies in flagellated or ciliated unicellular eukaryotic organisms, specifically Tetrahymena and Chlamydomonas. Reproducing these and other findings in vertebrates has required animal in vivo models. Zebrafish have fast become one of the primary organisms of choice for modeling vertebrate functional genetics. Rapid ex-utero development, proficient egg laying, ease of genetic manipulation, and affordability make zebrafish an attractive vertebrate research tool. Furthermore, zebrafish share over 80?% of disease causing genes with humans. In this article, we discuss the merits of using zebrafish to study BB functional genetics, review current knowledge of zebrafish BB ultrastructure and mechanisms of function, and consider the outlook for future zebrafish-based BB studies.
机译：对纤毛功能的研究大大地掩盖了对基体（BBs）在发育和疾病中的作用的了解。尽管这两个细胞器密切相关，但它们具有完成成功的细胞发育所需的特定作用。 BB的适当发育和功能是纤毛功能的基础。确实，影响纤毛发育的人类遗传疾病越来越多，统称为纤毛病。越来越多的证据表明，BB可以建立细胞极性，指导纤毛发生，并为纤毛轴突内所需的蛋白质提供停靠位点。鞭毛或纤毛单细胞真核生物，特别是四膜虫和衣原体的研究为我们对BB结构和功能的知识做出了重大贡献。在脊椎动物中复制这些发现和其他发现需要动物体内模型。斑马鱼已迅速成为建模脊椎动物功能遗传学的首选主要生物之一。快速的宫外发育，熟练的产卵，易于的基因操作和可负担性使斑马鱼成为有吸引力的脊椎动物研究工具。此外，斑马鱼占人类致病基因的80％以上。在本文中，我们讨论了使用斑马鱼研究BB功能遗传学的优点，回顾了斑马鱼BB超微结构和功能机制的当前知识，并考虑了基于斑马鱼的BB研究的前景。

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