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发现一种新的遗传性 RECQ 解旋酶疾病 RECON 综合征,将复制应激反应和基因组稳态定位为衰老和与年龄相关疾病的核心重要过程。

Discovery of a new hereditary RECQ helicase disorder RECON syndrome positions the replication stress response and genome homeostasis as centrally important processes in aging and age-related disease.

机构信息

Helicases and Genomic Integrity Section, Translational Gerontology Branch, National Institute on Aging, NIH, Baltimore, Maryland, USA.

Institute for Cancer and Genomic Sciences, University of Birmingham, Birmingham, United Kingdom.

出版信息

Ageing Res Rev. 2023 Apr;86:101887. doi: 10.1016/j.arr.2023.101887. Epub 2023 Feb 19.

Abstract

Characterizing the molecular deficiencies underlying human aging has been a formidable challenge as it is clear that a complex myriad of factors including genetic mutations, environmental influences, and lifestyle choices influence the deterioration responsible for human pathologies. In addition, the common denominators of human aging, exemplified by the newly updated hallmarks of aging (López-Otín et al., 2023), suggest multiple avenues and layers of crosstalk between pathways important for genome and cellular homeostasis, both of which are major determinants of both good health and lifespan. In this regard, we postulate that hereditary disorders characterized by chromosomal instability offer a unique window of insight into aging and age-related disease processes. Recently, we discovered a new RECQ helicase disorder, designated RECON syndrome attributed to bi-allelic mutations in the RECQL1 gene (Abu-Libdeh et al., 2022). Cells deficient in RECQL1 exhibit genomic instability and a compromised response to replication stress, providing further evidence for the significance of genome homeostasis to suppress disease phenotypes. Here we provide a perspective on the pathology of RECON syndrome to inform the reader as to how molecular defects in the RECQL1 gene contribute to underlying deficiencies in nucleic acid metabolism often seen in certain aging or age-related diseases.

摘要

阐明导致人类衰老的分子缺陷一直是一个艰巨的挑战,因为很明显,包括基因突变、环境影响和生活方式选择在内的一系列复杂因素都会影响导致人类疾病的恶化。此外,人类衰老的共同特征(如最近更新的衰老标志(López-Otín 等人,2023))表明,对于基因组和细胞内稳态都很重要的途径之间存在多种途径和层次的串扰,而这两者都是健康和寿命的主要决定因素。在这方面,我们假设以染色体不稳定性为特征的遗传性疾病为衰老和与年龄相关的疾病过程提供了一个独特的洞察窗口。最近,我们发现了一种新的 RECQ 解旋酶疾病,称为 RECON 综合征,归因于 RECQL1 基因的双等位基因突变(Abu-Libdeh 等人,2022)。缺乏 RECQL1 的细胞表现出基因组不稳定性和对复制应激的反应受损,这进一步证明了基因组内稳性对于抑制疾病表型的重要性。在这里,我们提供了对 RECON 综合征病理学的观点,以使读者了解 RECQL1 基因的分子缺陷如何导致某些衰老或与年龄相关的疾病中常见的核酸代谢缺陷。

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本文引用的文献

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Hallmarks of aging: An expanding universe.衰老的特征:一个不断扩大的领域。
Cell. 2023 Jan 19;186(2):243-278. doi: 10.1016/j.cell.2022.11.001. Epub 2023 Jan 3.
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