Ramesh V, Benoit L A, Crawford P, Harvey P T, Shows T B, Shih V E, Gusella J F
Neurology Service, Massachusetts General Hospital, Boston 02114.
Am J Hum Genet. 1988 Feb;42(2):365-72.
A cDNA probe (HOAT1) for ornithine aminotransferase (OAT) has recently been used to map (1) the structural gene for this enzyme to chromosome 10 and (2) several related DNA sequences to the X chromosome. We have defined six RFLPs for OAT, to explore its possible role in gyrate atrophy (GA) of the choroid and retina, an autosomal recessive genetic disorder associated with a deficiency of OAT activity. The RFLPs, which are detected by noncoding single-copy probes from the OAT gene and by subclones of the HOAT1 cDNA, all map on human chromosome 10, producing an overall level of heterozygosity for the OAT locus of 83%. Using the RFLPs, we have determined that the OAT locus segregates concordantly with GA in one available pedigree. Furthermore, the RFLPs display significant disequilibrium with GA, providing genetic evidence implicating a defect in the OAT structural gene as the cause of this disorder. The RFLPs for OAT are potentially applicable to prenatal diagnosis and carrier detection in families with a previous history of GA. They will also allow identification of specific haplotypes associated with GA chromosomes, as a guide for more detailed molecular-genetic investigations of the mutations underlying the disorder.
一种用于鸟氨酸转氨酶(OAT)的cDNA探针(HOAT1)最近被用于(1)将该酶的结构基因定位到10号染色体,以及(2)将几个相关的DNA序列定位到X染色体。我们已经确定了OAT的六个限制性片段长度多态性(RFLP),以探讨其在脉络膜和视网膜回旋性萎缩(GA)中的可能作用,GA是一种与OAT活性缺乏相关的常染色体隐性遗传病。这些RFLP通过来自OAT基因的非编码单拷贝探针和HOAT1 cDNA的亚克隆进行检测,全部定位在人类10号染色体上,OAT基因座的杂合性总体水平为83%。利用这些RFLP,我们在一个可用的家系中确定OAT基因座与GA共分离。此外,这些RFLP与GA表现出显著的不平衡,提供了遗传证据,表明OAT结构基因的缺陷是这种疾病的病因。OAT的RFLP可能适用于有GA病史家庭的产前诊断和携带者检测。它们还将允许识别与GA染色体相关的特定单倍型,作为对该疾病潜在突变进行更详细分子遗传学研究的指导。