Mutations Involved in Premature-Ageing Syndromes.

IF 2.6 Q2 GENETICS & HEREDITY Application of Clinical Genetics Pub Date : 2021-06-02 eCollection Date: 2021-01-01 DOI:10.2147/TACG.S273525
Fabio Coppedè
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引用次数: 12

Abstract

Premature-ageing syndromes are a heterogeneous group of rare genetic disorders resembling features of accelerated ageing and resulting from mutations in genes coding for proteins required for nuclear lamina architecture, DNA repair and maintenance of genome stability, mitochondrial function and other cellular processes. Hutchinson-Gilford progeria syndrome (HGPS) and Werner syndrome (WS) are two of the best-characterized progeroid syndromes referred to as childhood- and adulthood-progeria, respectively. This article provides an updated overview of the mutations leading to HGPS, WS, and to the spectrum of premature-ageing laminopathies ranging in severity from congenital restrictive dermopathy (RD) to adult-onset atypical WS, including RD-like laminopathies, typical and atypical HGPS, more and less severe forms of mandibuloacral dysplasia (MAD), Néstor-Guillermo progeria syndrome (NGPS), atypical WS, and atypical progeroid syndromes resembling features of HGPS and/or MAD but resulting from impaired DNA repair or mitochondrial functions, including mandibular hypoplasia, deafness, progeroid features, and lipodystrophy (MDPL) syndrome and mandibuloacral dysplasia associated to MTX2 (MADaM). The overlapping signs and symptoms among different premature-ageing syndromes, resulting from both a large genetic heterogeneity and shared pathological pathways underlying these conditions, require an expert clinical evaluation in specialized centers paralleled by next-generation sequencing of panels of genes involved in these disorders in order to establish as early as possible an accurate clinical and molecular diagnosis for a proper patient management.

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与过早衰老综合征相关的突变。
过早衰老综合征是一组异质性的罕见遗传疾病,类似于加速衰老的特征,由编码核层结构、DNA修复和维持基因组稳定性、线粒体功能和其他细胞过程所需蛋白质的基因突变引起。Hutchinson-Gilford早衰综合征(HGPS)和Werner综合征(WS)是两种最具特征的类早衰综合征,分别被称为儿童期和成年期早衰症。本文提供了导致HGPS、WS的突变的最新概述,以及从先天性限制性皮肤病(RD)到成人发病的非典型WS的严重程度不等的早衰层压板病谱,包括RD样层压板病、典型和非典型HGPS、更严重和不太严重的下颌关节发育不良(MAD)、n - guillermo早衰综合征(NGPS)、非典型WS、以及类似HGPS和/或MAD特征但由DNA修复或线粒体功能受损引起的非典型类早衰综合征,包括与MTX2 (MADaM)相关的下颌发育不全、耳聋、类早衰特征、脂肪营养不良(MDPL)综合征和下颌肢发育不良。不同早衰综合征之间的重叠体征和症状是由巨大的遗传异质性和共同的病理通路导致的,需要在专业中心进行专家临床评估,同时对与这些疾病有关的基因组进行下一代测序,以便尽早建立准确的临床和分子诊断,以便进行适当的患者管理。
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来源期刊
Application of Clinical Genetics
Application of Clinical Genetics Biochemistry, Genetics and Molecular Biology-Genetics
CiteScore
5.40
自引率
0.00%
发文量
20
审稿时长
16 weeks
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