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产后肌肉成熟的转录组学特征

Transcriptomic characterization of postnatal muscle maturation.

作者信息

Simon Alix, Djeddi Sarah, Bournon Pauline, Reiss David, Thompson Julie, Laporte Jocelyn

机构信息

Institut de Génétique et de Biologie Moléculaire et Cellulaire (IGBMC), CNRS UMR 7104, INSERM UMRS 1258, Université de Strasbourg, 67404 Illkirch, France.

Complex Systems and Translational Bioinformatics (CSTB), ICube laboratory - CNRS, Fédération de Médecine Translationnelle de Strasbourg (FMTS), Université de Strasbourg, 67000 Strasbourg, France.

出版信息

Dis Model Mech. 2025 Feb 1;18(2). doi: 10.1242/dmm.052098. Epub 2025 Mar 3.

Abstract

Gene differential expression and alternative splicing are mechanisms that give rise to a plethora of tissue-specific transcripts. Although these mechanisms have been studied in various tissues, their role during muscle maturation is not well understood. Because this stage of development is impaired in multiple muscular diseases, we used RNA sequencing to analyze transcriptome remodeling in skeletal muscle from late embryonic stage [embryonic day (E)18.5] to adult mice (7 weeks). Major transcriptomic changes were detected, especially in the first 2 weeks after birth, with a total of 8571 differentially expressed genes and 3096 alternatively spliced genes. Comparison of the two mechanisms showed that they regulate different biological processes essential for the structure and function of skeletal muscle. Investigation of genes mutated in muscle disorders revealed previously unknown transcripts. In particular, we validated a novel exon in Lrp4, a gene mutated in congenital myasthenia, in mice and humans. Overall, the characterization of the transcriptome in disease-relevant tissues revealed key pathways in the regulation of tissue maturation and function. Importantly, the exhaustive description of alternative splicing and resulting transcripts can improve genetic diagnosis of muscular diseases.

摘要

基因差异表达和可变剪接是产生大量组织特异性转录本的机制。尽管这些机制已在各种组织中得到研究,但其在肌肉成熟过程中的作用仍未得到充分理解。由于这一发育阶段在多种肌肉疾病中受损,我们使用RNA测序来分析从胚胎晚期[胚胎日(E)18.5]到成年小鼠(7周)骨骼肌中的转录组重塑。检测到主要的转录组变化,尤其是在出生后的前两周,共有8571个差异表达基因和3096个可变剪接基因。对这两种机制的比较表明,它们调节骨骼肌结构和功能所必需的不同生物学过程。对肌肉疾病中突变基因的研究揭示了以前未知的转录本。特别是,我们在小鼠和人类中验证了先天性肌无力中突变的基因Lrp4中的一个新外显子。总体而言,对疾病相关组织中转录组的表征揭示了组织成熟和功能调节中的关键途径。重要的是,对可变剪接和产生的转录本的详尽描述可以改善肌肉疾病的基因诊断。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/bad8/11911633/c36fbc7eb185/dmm-18-052098-g1.jpg

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