Identification of zebrafish insertional mutants with defects in visual system development and function
Identification of zebrafish insertional mutants with defects in visual system development and function
复制标题
DOI：
10.1534/genetics.104.039727
复制
发表时间：
2005-05-01
期刊：
影响因子：
3.3
通讯作者：
Dowling, JE
中科院分区：
文献类型：
--
作者：
Gross, JM;Perkins, BD;Dowling, JE
斑马鱼的遗传分析有助于识别视觉系统发育和功能所必需的基因。最近，一项大规模的逆转录病毒插入突变筛选完成，其中315个不同的基因发生突变，在受精后5天导致明显的表型缺陷。这些突变体中被破坏的基因已经被识别，这为研究单个器官系统的形成、功能或生理提供了独特的资源。为此，对该集合中的250个突变体进行了视觉系统突变体筛选，从组织学上检查每个突变体的眼睛形态学缺陷，从行为上检查整个视觉系统功能。发现了40个基因座，其破坏导致眼睛发育和/或视觉功能缺陷。突变体被分为以下表型类型，表现出以下缺陷：(1)形态发生，(2)生长和视网膜中央发育，(3)外周边缘区，(4)视网膜层压，(5)感光细胞层，(6)视网膜色素上皮，(7)晶状体，(8)视网膜包膜，(9)行为。这些突变体中受影响的基因突出了脊椎动物视觉系统的发育、维持和功能所必需的一组不同的蛋白质。
Genetic analysis in zebrafish has been instrumental in identifying genes necessary for visual system development and function. Recently, a large-scale retroviral insertional mutagenesis screen, in which 315 different genes were mutated, that resulted in obvious phenotypic defects by 5 days postfertilization was completed. That the disrupted gene has been identified in each of these mutants provides unique resource through which the formation, function, or physiology of individual organ systems can be studied. To that end, a screen for visual system mutants was performed on 250 of the mutants in this collection, examining each of them histologically for morphological defects in the eye and behaviorally for overall visual system function. Forty loci whose disruption resulted in defects in eye development and/or visual function were identified. The mutants have been divided into the following phenotypic classes that show defects in: (1) morphogenesis, (2) growth and central retinal development, (3) the peripheral marginal zone, (4) retinal lamination, (5) the photoreceptor cell layer, (6) the retinal pigment epithelium, (7) the lens, (8) retinal containment, and (9) behavior. The affected genes in these mutants highlight a diverse set of proteins necessary for the development, maintenance, and function of the vertebrate visual system.