Sobczak Krzysztof, Krzyzosiak Wlodzimierz J
Laboratory of Cancer Genetics, Institute of Bioorganic Chemistry, Polish Academy of Sciences, Noskowskiego 12/14, 61-704 Poznan, Poland.
J Biol Chem. 2005 Feb 4;280(5):3898-910. doi: 10.1074/jbc.M409984200. Epub 2004 Nov 8.
Spinocerebellar ataxia type 2 (SCA2), one of the hereditary human neurodegenerative disorders, is caused by the expansion of the CAG tandem repeats in the translated sequence of the SCA2 gene. In a normal population the CAG repeat is polymorphic not only in length but also in the number and localization of its CAA interruptions. The aim of this study was to determine the structure of the repeat region in the normal and mutant SCA2 transcripts and to reveal the structural basis of its normal function and dysfunction. We show here that the properties of the CAA interruptions are major determinants of the CAG repeat folding in the normal SCA2 transcripts. We also show that the uninterrupted repeats in mutant transcripts form slippery hairpins, whose length is further reduced by the base pairing of the repeat portion with a specific flanking sequence. The structural organization of the repeat interruption systems present in other human transcripts, such as SCA1, TBP, FOXP2, and MAML2, are also discussed.
2型脊髓小脑共济失调(SCA2)是人类遗传性神经退行性疾病之一,由SCA2基因翻译序列中CAG串联重复序列的扩增引起。在正常人群中,CAG重复序列不仅长度具有多态性,其CAA中断的数量和定位也具有多态性。本研究的目的是确定正常和突变型SCA2转录本中重复区域的结构,并揭示其正常功能和功能障碍的结构基础。我们在此表明,CAA中断的特性是正常SCA2转录本中CAG重复序列折叠的主要决定因素。我们还表明,突变型转录本中的不间断重复序列形成滑动发夹结构,其长度会因重复部分与特定侧翼序列的碱基配对而进一步缩短。本文还讨论了其他人类转录本(如SCA1、TBP、FOXP2和MAML2)中存在的重复中断系统的结构组织。