Dipartimento di Scienze Neurologiche, Neuropsicologiche, Morfologiche e Motorie, Sezione di Anatomia e Istologia, Università degli Studi di Verona, Strada Le Grazie 8, Verona, Italy.
Eur J Histochem. 2012 Aug 10;56(3):e36. doi: 10.4081/ejh.2012.e36.
In the cell nucleus, the gene primary transcripts undergo molecular processing to generate mature RNAs, which are finally exported to the cytoplasm. These mRNA maturation events are chronologically and spatially ordered, and mostly occur on distinct ribonucleoprotein (RNP)-containing structures. Defects in the mRNA maturation pathways have been demonstrated in myotonic dystrophy type 1 (DM1) and type 2 (DM2) whose characteristic multisystemic features are caused by the expansion of two distinct nucleotide sequences: (CTG)n in the DMPK gene on chromosome 19q13 in DM1, and (CCTG)n in the ZNF9 gene on chromosome 3q21 in DM2. By combining biomolecular and cytochemical techniques, it has been shown that the basic mechanisms of DMs reside in the accumulation of CUG- or CCUG-containing transcripts in intranuclear foci where several RNA-binding proteins necessary for the physiological processing of pre-mRNA are sequestered. Moreover, a nucleoplasmic accumulation of splicing and cleavage factors has been found in DMs. This suggests that the dystrophic phenotype could depend on a general alteration of the pre-mRNA post-transcriptional pathway. Interestingly, the accumulation of pre-mRNA processing factors in the myonuclei of DM1 and DM2 patients is reminiscent of the nuclear alterations typical of sarcopenia, i.e., the loss of muscle mass and function which physiologically occurs during ageing. Consistently, in an in vitro study, we observed that satellite-cell-derived DM2 myoblasts show cell senescence alterations and impairment of the pre-mRNA maturation pathways earlier than the myoblasts from healthy patient. These results suggest possible common cellular mechanisms responsible for skeletal muscle wasting in sarcopenia and in myotonic dystrophy.
在细胞核中,基因的初级转录本经历分子加工,生成成熟的 RNA,最终被输出到细胞质。这些 mRNA 成熟事件在时间和空间上是有序的,主要发生在不同的核糖核蛋白 (RNP) 包含的结构上。肌强直性营养不良 1 型 (DM1) 和 2 型 (DM2) 的 mRNA 成熟途径缺陷已被证明,其特征性多系统特征是由两个不同核苷酸序列的扩展引起的:DM1 中 19q13 染色体上 DMPK 基因中的 (CTG)n,以及 DM2 中 3q21 染色体上 ZNF9 基因中的 (CCTG)n。通过结合生物分子和细胞化学技术,已经表明 DMs 的基本机制在于 CUG-或 CCUG-含有转录物在核内焦点中的积累,其中几种 RNA 结合蛋白对于 pre-mRNA 的生理加工是必需的。此外,在 DMs 中发现了剪接和切割因子的核质积累。这表明,营养不良表型可能依赖于 pre-mRNA 转录后途径的普遍改变。有趣的是,DM1 和 DM2 患者的肌核中 pre-mRNA 加工因子的积累让人联想到与骨骼肌减少症相关的核改变,即肌肉质量和功能的生理性丧失随着年龄的增长而发生。一致地,在一项体外研究中,我们观察到卫星细胞衍生的 DM2 成肌细胞表现出细胞衰老改变和 pre-mRNA 成熟途径的损伤,早于来自健康患者的成肌细胞。这些结果表明,骨骼肌减少症和肌强直性营养不良中可能存在导致骨骼肌消耗的共同细胞机制。