Matsuura Tohru, Fang Ping, Pearson Christopher E, Jayakar Parul, Ashizawa Tetsuo, Roa Benjamin B, Nelson David L
Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA.
Am J Hum Genet. 2006 Jan;78(1):125-9. doi: 10.1086/498654. Epub 2005 Nov 15.
Spinocerebellar ataxia type 10 (SCA10) is one of numerous genetic disorders that result from simple repeat expansions. SCA10 is caused by expansion of an intronic ATTCT pentanucleotide repeat tract. It is clinically characterized by progressive ataxia, seizures, and anticipation, which can vary within and between families. We report two SCA10 families showing distinct frequencies of seizures and correlations of repeat length with age at onset. One family displayed uninterrupted ATTCT expansions, whereas the other showed multiple interruptions of the repeat by nonconsensus repeat units, which differed both in the length and/or sequence of the repeat unit. Disease-causing microsatellite expansions have been assumed to be composed of uninterrupted pure repeats. Our findings for SCA10 challenge this convention and suggest that the purity of the expanded repeat element may be a disease modifier.
10型脊髓小脑共济失调(SCA10)是由简单重复序列扩增导致的众多遗传性疾病之一。SCA10是由内含子ATTCT五核苷酸重复序列的扩增引起的。其临床特征为进行性共济失调、癫痫发作和遗传早现,这些症状在家族内部和家族之间可能有所不同。我们报告了两个SCA10家族,它们表现出不同的癫痫发作频率以及重复序列长度与发病年龄的相关性。一个家族显示出不间断的ATTCT扩增,而另一个家族则显示该重复序列被非一致性重复单元多次打断,这些非一致性重复单元在重复单元的长度和/或序列上均有所不同。致病微卫星扩增一直被认为是由不间断的纯重复序列组成。我们对SCA10的研究结果对这一传统观念提出了挑战,并表明扩增重复元件的纯度可能是一种疾病修饰因子。