McClatchey A I, Kaufman D L, Berson E L, Tobin A J, Shih V E, Gusella J F, Ramesh V
Neurogenetics Laboratory, Massachusetts General Hospital East, Charlestown 02129.
Am J Hum Genet. 1990 Nov;47(5):790-4.
Gyrate atrophy (GA), a recessive eye disease involving progressive vision loss due to chorioretinal degeneration, is associated with the deficiency of the mitochondrial enzyme ornithine aminotransferase (OAT), with consequent hyperornithinemia. We and others have reported a number of missense mutations at the OAT locus which result in GA. Here we report a GA patient of Danish/Swedish ancestry in whom one OAT allele produces an mRNA that is missing a single 96-bp exon relative to the normal mRNA. Polymerase-chain-reaction amplification and sequencing revealed a 9-bp deletion covering the splice acceptor region of exon 5, resulting in the absence of exon 5 sequences from the mRNA with no disruption to the reading frame. This mutation, which was not present in 15 other independent GA patients, adds to the array of allelic heterogeneity observed in GA and represents the first example of a splicing mutation associated with this disorder.
回旋状萎缩(GA)是一种隐性眼病,由于脉络膜视网膜变性导致进行性视力丧失,与线粒体酶鸟氨酸转氨酶(OAT)缺乏及随之而来的高鸟氨酸血症有关。我们和其他人已经报道了OAT基因座上的一些错义突变,这些突变会导致GA。在此,我们报告一名具有丹麦/瑞典血统的GA患者,其一个OAT等位基因产生的mRNA相对于正常mRNA缺失一个96bp的外显子。聚合酶链反应扩增和测序显示,一个9bp的缺失覆盖了外显子5的剪接受体区域,导致mRNA中没有外显子5序列,但阅读框未被破坏。该突变在其他15名独立的GA患者中未出现,增加了GA中观察到的等位基因异质性,并且代表了与该疾病相关的剪接突变的首个例子。