ENU-induced late-onset night blindness associated with rod photoreceptor cell degeneration in zebrafish
ENU-induced late-onset night blindness associated with rod photoreceptor cell degeneration in zebrafish
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DOI：
10.1016/j.mad.2003.08.004
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发表时间：
2003-12-01
影响因子：
5.3
通讯作者：
Li, L
中科院分区：
文献类型：
--
作者：
Maaswinkel, H;Mason, B;Li, L
我们描述了一个显性突变，夜盲d（nbd），导致晚发性视杆细胞变性在斑马鱼。用N-乙基-N-亚硝基脲（ENU）处理雄性斑马鱼诱导突变。使用基于视觉介导的逃避反应的行为测定来测试视觉灵敏度。在年轻的时候，杂合（nbd（+/-））鱼没有表现出任何夜盲症或视网膜变性的迹象。在2岁时，他们的行为评估的视觉灵敏度下降，虽然没有改变视网膜电图（ERG）检测。组织学显示，在突变体视网膜中，视杆细胞外节（ROS）变薄。在纯合子幼虫（nbd（-/-））中，在受精后约2天（dpf）可检测到大量神经变性。它们很早就去世了，通常不晚于8 dpf。总之，nbd是一种显性突变，导致晚发性夜盲症，进展缓慢。然而，从纯合子幼虫的系统功能障碍中可以清楚地看出，nbd并不是感光细胞特异性的。(C)2003年由Elsevier爱尔兰有限公司出版
We describe a dominant mutation, night blindness d (nbd), that causes late-onset rod photoreceptor cell degeneration in zebrafish. The mutation was induced by treating male zebrafish with N-ethyl-N-nitrosourea (ENU). Visual sensitivity was tested using a behavioral assay based on a visually mediated escape response. At a young age, the heterozygous (nbd(+/-)) fish did not show any signs of night blindness or retinal degeneration. At 2 years, their behaviorally assessed visual sensitivity was decreased, albeit no alterations in the electroretinogram (ERG) were detected. Histology revealed that in the mutant retinas the rod photoreceptor cell outer segments (ROS) were thinned out. In homozygous larvae (nbd(-/-)), mass neural degeneration was detectable at about 2 days post fertilization (dpf). They died at an early age, usually no later than 8 dpf. In conclusion, nbd is a dominant mutation that causes late-onset night blindness with slow progression. However, nbd is not photoreceptor cell-specific, as becomes clear from the systemic dysfunctions of the homozygous larvae. (C) 2003 Published by Elsevier Ireland Ltd.