端粒生物学疾病:从先天性角化障碍到其他疾病。

IF 3.6 4区 医学 Q1 MEDICINE, GENERAL & INTERNAL Postgraduate Medical Journal Pub Date : 2024-08-29 DOI:10.1093/postmj/qgae102
Kleoniki Roka, Elena Solomou, Antonis Kattamis, Eftychia Stiakaki
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引用次数: 0

摘要

端粒酶功能或端粒维持功能缺陷会导致基因组不稳定。端粒长度和/或损耗的改变是被称为端粒生物学疾病或端粒病的罕见疾病的主要特征。这些疾病的分子基础和评估端粒长度的前沿方法的最新进展加深了我们对这一主题的理解。即使是同一家族中的携带者,也有多器官表现和不同表型的报道。在这种情况下,除了先天性角化不良外,以前被认为是特发性的疾病(如肺纤维化、肝硬化)经常与潜在的端粒维持机制缺陷有关。此外,这些患者容易罹患特定类型的癌症,并对标准化疗方案表现出特殊的敏感性和毒性。本综述描述了端粒生物学紊乱在儿童和成人患者中的各种临床表现、其与致病变异的相关性,以及在治疗过程中的注意事项,以提高人们的认识并改进多学科方法。
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Telomere biology disorders: from dyskeratosis congenita and beyond.

Defective telomerase function or telomere maintenance causes genomic instability. Alterations in telomere length and/or attrition are the primary features of rare diseases known as telomere biology disorders or telomeropathies. Recent advances in the molecular basis of these disorders and cutting-edge methods assessing telomere length have increased our understanding of this topic. Multiorgan manifestations and different phenotypes have been reported even in carriers within the same family. In this context, apart from dyskeratosis congenita, disorders formerly considered idiopathic (i.e. pulmonary fibrosis, liver cirrhosis) frequently correlate with underlying defective telomere maintenance mechanisms. Moreover, these patients are prone to developing specific cancer types and exhibit exceptional sensitivity and toxicity in standard chemotherapy regimens. The current review describes the diverse spectrum of clinical manifestations of telomere biology disorders in pediatric and adult patients, their correlation with pathogenic variants, and considerations during their management to increase awareness and improve a multidisciplinary approach.

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来源期刊
Postgraduate Medical Journal
Postgraduate Medical Journal 医学-医学:内科
CiteScore
8.50
自引率
2.00%
发文量
131
审稿时长
2.5 months
期刊介绍: Postgraduate Medical Journal is a peer reviewed journal published on behalf of the Fellowship of Postgraduate Medicine. The journal aims to support junior doctors and their teachers and contribute to the continuing professional development of all doctors by publishing papers on a wide range of topics relevant to the practicing clinician and teacher. Papers published in PMJ include those that focus on core competencies; that describe current practice and new developments in all branches of medicine; that describe relevance and impact of translational research on clinical practice; that provide background relevant to examinations; and papers on medical education and medical education research. PMJ supports CPD by providing the opportunity for doctors to publish many types of articles including original clinical research; reviews; quality improvement reports; editorials, and correspondence on clinical matters.
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