LeProust E M, Pearson C E, Sinden R R, Gao X
Department of Chemistry, University of Houston, Houston, TX 77204-5641, USA.
J Mol Biol. 2000 Oct 6;302(5):1063-80. doi: 10.1006/jmbi.2000.4073.
The onset and progress of Friedreich's ataxia (FRDA) is associated with the genetic instability of the (GAA).(TTC) trinucleotide repeats located within the frataxin gene. The instability of these repeats may involve the formation of an alternative DNA structure. Poly-purine (R)/poly-pyrimidine (Y) sequences typically form triplex DNA structures which may contribute to genetic instability. Conventional wisdom suggested that triplex structures formed by these poly-purine (R)/poly-pyrimidine (Y) sequences may contribute to their genetic instability. Here, we report the characterization of the single-stranded GAA and TTC sequences and their mixtures using NMR, UV-melting, and gel electrophoresis, as well as chemical and enzymatic probing methods. We show that the FRDA GAA/TTC, repeats are capable of forming various alternative structures. The most intriguing is the observation of a parallel (GAA).(TTC) duplex in equilibrium with the antiparallel Watson-Crick (GAA).(TTC) duplex. We also show that the GAA strands form self-assembled structures, whereas the TTC strands are essentially unstructured. Finally, we demonstrate that the FRDA repeats form only the YRY triplex (but not the RRY triplex) at neutral pH and the complete formation of the YRY triplex requires the ratio of GAA to TTC strand larger than 1:2. The structural features presented here and in other studies distinguish the FRDA (GAA)¿(TTC) repeats from the fragile X (CGG).CCG), myotonic dystrophy (CTG).(CAG) and the Huntington (CAG).(CTG) repeats.
弗里德赖希共济失调(FRDA)的发病和进展与位于铁调素基因内的(GAA)·(TTC)三核苷酸重复序列的遗传不稳定性有关。这些重复序列的不稳定性可能涉及形成一种替代的DNA结构。多嘌呤(R)/多嘧啶(Y)序列通常形成三链DNA结构,这可能导致遗传不稳定性。传统观点认为,这些多嘌呤(R)/多嘧啶(Y)序列形成的三链结构可能导致其遗传不稳定性。在此,我们报告了使用核磁共振(NMR)、紫外熔解和凝胶电泳以及化学和酶促探测方法对单链GAA和TTC序列及其混合物的表征。我们表明,FRDA的GAA/TTC重复序列能够形成各种替代结构。最引人注目的是观察到平行的(GAA)·(TTC)双链体与反平行的沃森-克里克(GAA)·(TTC)双链体处于平衡状态。我们还表明,GAA链形成自组装结构,而TTC链基本上是无结构的。最后,我们证明,在中性pH条件下,FRDA重复序列仅形成YRY三链体(而非RRY三链体),并且YRY三链体的完全形成需要GAA与TTC链的比例大于1:2。本文及其他研究中呈现的结构特征将FRDA的(GAA)·(TTC)重复序列与脆性X综合征的(CGG)·(CCG)、强直性肌营养不良的(CTG)·(CAG)以及亨廷顿舞蹈症的(CAG)·(CTG)重复序列区分开来。