Feng Y, Zhang F, Lokey L K, Chastain J L, Lakkis L, Eberhart D, Warren S T
Howard Hughes Medical Institute, Emory University School of Medicine, Atlanta, GA 30322.
Science. 1995 May 5;268(5211):731-4. doi: 10.1126/science.7732383.
Fragile X syndrome is the result of the unstable expansion of a trinucleotide repeat in the 5'-untranslated region of the FMR1 gene. Fibroblast subclones from a mildly affected patient, each containing stable FMR1 alleles with 57 to 285 CGG repeats, were shown to exhibit normal steady-state levels of FMR1 messenger RNA. However, FMR protein was markedly diminished from transcript with more than 200 repeats. Such transcripts were associated with stalled 40S ribosomal subunits. These results suggest that a structural RNA transition beyond 200 repeats impedes the linear 40S migration along the 5'-untranslated region. This results in translational inhibition by trinucleotide repeat expansion.
脆性X综合征是FMR1基因5'-非翻译区三核苷酸重复序列不稳定扩增的结果。来自一名轻度受影响患者的成纤维细胞亚克隆,每个亚克隆都含有稳定的FMR1等位基因,具有57至285个CGG重复序列,显示出FMR1信使RNA的正常稳态水平。然而,FMR蛋白在重复序列超过200个的转录本中明显减少。这些转录本与停滞的40S核糖体亚基相关。这些结果表明,超过200个重复序列的结构RNA转变阻碍了40S沿着5'-非翻译区的线性迁移。这导致了三核苷酸重复序列扩增引起的翻译抑制。