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首页> 外文期刊>The European Journal of Neuroscience >Retinal function and morphology in two zebrafish models of oculo-renal syndromes.
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Retinal function and morphology in two zebrafish models of oculo-renal syndromes.

机译：两种眼-肾综合征的斑马鱼模型的视网膜功能和形态。

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摘要

We characterized visual system defects in two recessive zebrafish mutants oval and elipsa. These mutants share the syndromic phenotype of outer retinal dystrophy in conjunction with cystic renal disorder. We tested the function of the larval visual system in a behavioural assay, eliciting optokinetic eye movements by high-contrast motion stimulation while recording eye movements in parallel. Visual stimulation did not elicit eye movements in mutant larvae, while spontaneous eye movements could be observed. The retina proved to be unresponsive to light using electroretinography, indicative of a defect in the outer retina. Histological analysis of mutant retinas revealed progressive degeneration of photoreceptors, initiated in central retinal locations and spreading to more peripheral regions with increasing age. The inner retina remains unaffected by the mutation. Photoreceptors display cell type-specific immunoreactivity prior to apoptotic cell death, arguing for a dystrophic defect. Genomic mapping employing simple sequence-length polymorphisms located both mutations on different regions of zebrafish linkage group 9. These mutants may serve as accessible animal models of human outer retinal dystrophies, including oculo-renal diseases, and show the general usefulness of a behavioural genetic approach to study visual system development in the model vertebrate zebrafish.
机译：我们表征了两个隐性斑马鱼突变体椭圆形和椭圆形的视觉系统缺陷。这些突变体与囊性肾病有共同的外部视网膜营养不良的症状表型。我们在行为分析中测试了幼虫视觉系统的功能，通过高对比度运动刺激引发了视动眼运动，同时记录了眼动。视觉刺激未引起突变幼虫的眼球运动，而可以观察到自发眼球运动。视网膜电图证明视网膜对光无反应，表明视网膜外层缺损。突变视网膜的组织学分析表明，感光细胞逐渐退化，始于视网膜中央部位，并随着年龄的增长而扩散到更多的周边区域。内视网膜仍不受突变影响。感光细胞在凋亡细胞死亡之前显示出细胞类型特异性免疫反应性，这是营养不良的原因。使用简单的序列长度多态性进行基因组定位，将两个突变都定位在斑马鱼连锁组9的不同区域。这些突变体可作为人类外部视网膜营养不良（包括眼-肾疾病）的可访问动物模型，并显示出行为遗传方法的一般有用性研究模型斑马鱼的视觉系统发育。

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