Identification of biallelic POLA2 variants in two families with an autosomal recessive telomere biology disorder

IF 4.6 2区 生物学 Q2 BIOCHEMISTRY & MOLECULAR BIOLOGY European Journal of Human Genetics Pub Date : 2024-11-30 DOI:10.1038/s41431-024-01722-8
Malin Kvarnung, Maria Pettersson, Pattra Chun-on, Maryam Rafati, Lisa J. McReynolds, Anna Norberg, Pedro Luis Moura, Ida Pesonen, Roza Chaireti, Boa Grönros Söderholm, Julia Burlin, Jenny Rydén, Eva Hellström Lindberg, Neelam Giri, Sharon A. Savage, Suneet Agarwal, Ann Nordgren, Bianca Tesi
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Abstract

POLA2 encodes the accessory subunit of DNA polymerase α (polα)/primase, which is crucial for telomere C-strand fill-in. Incomplete fill-in of the C-rich telomeric strand after DNA replication has been proposed as a mechanism for Coats plus syndrome, a phenotype within the broader spectrum of telomere biology disorders (TBD). Coats plus syndrome has so far been associated with pathogenic variants in POT1, CTC1, and STN1. Here we report the findings of biallelic deleterious rare variants in POLA2 gene detected by whole genome sequencing and segregation analysis in five young adults from two unrelated families. All five individuals displayed abnormally short telomeres and a clinical phenotype suggesting a TBD disorder with Coats plus features including retinal and gastrointestinal telangiectasias. Our results suggest POLA2 as a novel autosomal recessive gene for a TBD with Coats plus features.

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两个常染色体隐性端粒生物学疾病家族的双等位基因POLA2变异的鉴定。
POLA2编码DNA聚合酶α (polα)/引物酶的辅助亚基,对端粒c链的填充至关重要。DNA复制后富c端粒链的不完全填充被认为是Coats +综合征的一种机制,这是端粒生物学疾病(TBD)的一种更广泛的表型。迄今为止,Coats +综合征与POT1、CTC1和STN1的致病变异有关。在这里,我们报告了通过全基因组测序和分离分析在来自两个不相关家庭的5名年轻人中检测到POLA2基因双等位基因有害罕见变异的发现。所有五个人都表现出异常短的端粒和临床表型,表明TBD疾病具有Coats +特征,包括视网膜和胃肠道毛细血管扩张。我们的研究结果表明POLA2是具有Coats +特征的TBD的一种新的常染色体隐性基因。
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来源期刊
European Journal of Human Genetics
European Journal of Human Genetics 生物-生化与分子生物学
CiteScore
9.90
自引率
5.80%
发文量
216
审稿时长
2 months
期刊介绍: The European Journal of Human Genetics is the official journal of the European Society of Human Genetics, publishing high-quality, original research papers, short reports and reviews in the rapidly expanding field of human genetics and genomics. It covers molecular, clinical and cytogenetics, interfacing between advanced biomedical research and the clinician, and bridging the great diversity of facilities, resources and viewpoints in the genetics community. Key areas include: -Monogenic and multifactorial disorders -Development and malformation -Hereditary cancer -Medical Genomics -Gene mapping and functional studies -Genotype-phenotype correlations -Genetic variation and genome diversity -Statistical and computational genetics -Bioinformatics -Advances in diagnostics -Therapy and prevention -Animal models -Genetic services -Community genetics
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