Varani L, Hasegawa M, Spillantini M G, Smith M J, Murrell J R, Ghetti B, Klug A, Goedert M, Varani G
Medical Research Council Laboratory of Molecular Biology, Hills Road, Cambridge CB2 2QH, United Kingdom.
Proc Natl Acad Sci U S A. 1999 Jul 6;96(14):8229-34. doi: 10.1073/pnas.96.14.8229.
Coding region and intronic mutations in the tau gene cause frontotemporal dementia and parkinsonism linked to chromosome 17. Intronic mutations and some missense mutations increase splicing in of exon 10, leading to an increased ratio of four-repeat to three-repeat tau isoforms. Secondary structure predictions have led to the proposal that intronic mutations and one missense mutation destabilize a putative RNA stem-loop structure located close to the splice-donor site of the intron after exon 10. We have determined the three-dimensional structure of this tau exon 10 splicing regulatory element RNA by NMR spectroscopy. We show that it forms a stable, folded stem-loop structure whose thermodynamic stability is reduced by frontotemporal dementia and parkinsonism linked to chromosome 17 mutations and increased by compensatory mutations. By exon trapping, the reduction in thermodynamic stability is correlated with increased splicing in of exon 10.
tau基因中的编码区和内含子突变会导致与17号染色体相关的额颞叶痴呆和帕金森综合征。内含子突变和一些错义突变会增加外显子10的剪接,导致四重复tau异构体与三重复tau异构体的比例增加。二级结构预测表明,内含子突变和一个错义突变会破坏位于外显子10后内含子剪接供体位点附近的假定RNA茎环结构的稳定性。我们通过核磁共振光谱法确定了这种tau外显子10剪接调控元件RNA的三维结构。我们发现它形成了一个稳定的折叠茎环结构,其热力学稳定性因与17号染色体相关的额颞叶痴呆和帕金森综合征突变而降低,并因补偿性突变而增加。通过外显子捕获,热力学稳定性的降低与外显子10剪接的增加相关。