Rudolph J A, Spier S J, Byrns G, Hoffman E P
Department of Molecular Genetics & Biochemistry, University of Pittsburgh School of Medicine, Pennsylvania 15261.
Anim Genet. 1992;23(3):241-50. doi: 10.1111/j.1365-2052.1992.tb00136.x.
A genetic disease observed in certain Quarter horses is hyperkalaemic periodic paralysis (HYPP). This disease causes attacks of paralysis which can be induced by ingestion of potassium. Recent studies have shown that HYPP in humans is due to single base changes within the adult skeletal muscle sodium channel gene. A large Quarter horse pedigree segregating dominant HYPP was studied to determine if mutations of the sodium channel gene are similarly responsible for HYPP in horses. We used cross-species, PCR-mediated, cDNA cloning and sequencing of the horse adult skeletal muscle sodium channel alpha-subunit gene to identify a polymorphism, and then used this polymorphism to see if the horse sodium channel gene was genetically linked to HYPP in horses. The sodium channel gene was indeed found to be tightly linked to HYPP (LOD = 2.7, theta = 0). Our results suggest that HYPP in horses involves the same gene as the clinically similar human disease, and indicates that these are homologous disorders. The future identification of the specific sodium channel mutation causing HYPP in Quarter horses will permit the development of accurate molecular diagnostics of this condition, as has been recently shown for humans.
在某些夸特马中观察到的一种遗传性疾病是高钾性周期性麻痹(HYPP)。这种疾病会引发麻痹发作,摄入钾可诱发发作。最近的研究表明,人类的HYPP是由于成人骨骼肌钠通道基因内的单碱基变化所致。对一个分离显性HYPP的大型夸特马谱系进行了研究,以确定钠通道基因的突变是否同样导致马匹的HYPP。我们使用跨物种、PCR介导的马成人骨骼肌钠通道α亚基基因的cDNA克隆和测序来鉴定一种多态性,然后利用这种多态性来观察马钠通道基因是否与马匹的HYPP存在遗传连锁。确实发现钠通道基因与HYPP紧密连锁(对数优势比分=2.7,重组率=0)。我们的结果表明,马匹的HYPP涉及与临床上类似的人类疾病相同的基因,这表明这些是同源性疾病。未来确定导致夸特马HYPP的特定钠通道突变,将能够开发出针对这种病症的准确分子诊断方法,就像最近在人类中所显示的那样。