Bowes C, Danciger M, Kozak C A, Farber D B
Jules Stein Eye Institute, University of California, Los Angeles 90024.
Proc Natl Acad Sci U S A. 1989 Dec;86(24):9722-6. doi: 10.1073/pnas.86.24.9722.
The inherited retinal degeneration of the rd mouse results in the exclusive loss of one cell type, the photoreceptors. We took advantage of this visual-cell loss to devise a strategy for the isolation of photoreceptor-specific cDNAs based on the use of subtractive and differential hybridizations. The resulting pool of photoreceptor-specific cDNAs was screened for a candidate cDNA for the rd gene, and a putative rd cDNA that maps to mouse chromosome 5, the chromosome to which the rd gene has been assigned, was identified. On Northern blots the candidate rd cDNA hybridizes a 3.3-kilobase RNA species from 9- to 11-day-old developing normal retina and, much more faintly, a 3.6-kb RNA species from age-matched rd retina. The 0.3-kilobase difference in the size of the mRNAs hybridized suggests that a structural alteration in the gene corresponding to the candidate rd cDNA has occurred in the rd mouse. This was further supported by the detection of polymorphisms between rd/rd and +/+ mouse genomic DNA after digestion with restriction endonucleases and probing with the candidate rd cDNA. Expression of mRNAs hybridized by the candidate rd cDNA is detected in normal and diseased retinas at postnatal day 1 but the signal intensity is considerably lower in the rd retina. To our knowledge, this is the earliest molecular defect reported in the rd retina that is observed prior to any phenotypic signs of photoreceptor degeneration.
rd小鼠的遗传性视网膜变性导致一种细胞类型——光感受器——的特异性缺失。我们利用这种视觉细胞的缺失,设计了一种基于消减杂交和差异杂交来分离光感受器特异性cDNA的策略。对所得的光感受器特异性cDNA文库进行筛选,以寻找rd基因的候选cDNA,结果鉴定出一个推定的rd cDNA,它定位于小鼠5号染色体,即rd基因所在的染色体。在Northern印迹上,候选rd cDNA与来自9至11日龄正常发育视网膜的一种3.3千碱基RNA杂交,与来自同龄rd视网膜的一种3.6千碱基RNA杂交则要微弱得多。杂交mRNA大小上0.3千碱基的差异表明,在rd小鼠中,与候选rd cDNA相对应的基因发生了结构改变。在用限制性内切酶消化并用候选rd cDNA进行探针杂交后,在rd/rd和+/+小鼠基因组DNA之间检测到多态性,这进一步支持了上述结论。在出生后第1天,在正常和患病视网膜中均检测到与候选rd cDNA杂交的mRNA的表达,但在rd视网膜中的信号强度要低得多。据我们所知,这是在rd视网膜中报道的最早的分子缺陷,它在光感受器变性的任何表型迹象出现之前就已观察到。