全外显子组测序鉴定了来自巴基斯坦近亲家庭的无精子症患者的罕见隐性变异。

IF 2.3 3区 生物学 Q3 BIOCHEMISTRY & MOLECULAR BIOLOGY Molecular Genetics and Genomics Pub Date : 2024-12-03 DOI:10.1007/s00438-024-02205-7
Islam Uddin, Iqra Zafar, Caoling Xu, Wenqing Li, Muhammad Imran Khan, Limin Wu, Jianqiang Bao
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引用次数: 0

摘要

无精子症是一种严重的男性不育症,其特征是射精中完全没有精子,约占男性人口的1%,大多数病例归因于配子体发育失败引起的非阻塞性无精子症(NOA)。NOA有多种遗传起源,包括染色体异常、Y染色体微缺失和单基因突变。尽管全外显子组测序(WES)已经确定了30多个与NOA相关的候选基因,但大多数病例的遗传原因尚未阐明。在我们的研究中,我们从巴基斯坦农村地区招募的21个男性不育家庭中选择了7个诊断为无精子症的近亲家庭。收集患者和可生育对照组的血液样本进行DNA提取,然后进行WES鉴定与男性不育相关的潜在致病隐性单基因变异。我们成功鉴定了7个家族中5个家族的5个有害变异,包括WWC2、RPL10L和SOHLH1的3个错义双等位基因替换,ESX1的一个半合子缺失和TXNDC2的纯合子缺失,它们与人类男性不育的无精子症具有潜在的致病相关性。这些新发现增强了我们对无精子症复杂病因的分子机制的理解,为遗传咨询和诊断提供了有价值的见解,并为未来的治疗方法铺平了道路。
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Whole-exome sequencing identifies rare recessive variants in azoospermia patients from consanguineous Pakistani families.

Azoospermia, a severe form of male infertility characterized by the complete absence of sperm in the ejaculate, affects about 1% of the male population, with most cases attributed to nonobstructive azoospermia (NOA) caused by gametogenic failure. NOA has various genetic origins, including chromosomal abnormalities, Y chromosome microdeletions, and monogenic mutations. Although whole-exome sequencing (WES) has identified over thirty candidate genes associated with NOA, the genetic causes of most cases have yet to be elucidated. In our study, we selected seven consanguineous families diagnosed with azoospermia from a total of 21 male infertile families recruited from the rural area of Pakistan. Blood samples were collected from both patients and fertile controls for DNA extraction, followed by WES to identify potential causative recessive monogenic variants linked to male infertility. We successfully identified five deleterious variants among five of the seven families, including three missense biallelic substitutions in WWC2, RPL10L, and SOHLH1, a hemizygous deletion in ESX1, and a homozygous deletion in TXNDC2, which have potentially pathogenic relevance to the azoospermia of human male infertility. These novel findings enhance our understanding of the molecular mechanisms underlying the complex etiology of azoospermia, offering valuable insights for genetic counseling and diagnostics and paving the way for future therapeutic approaches.

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来源期刊
Molecular Genetics and Genomics
Molecular Genetics and Genomics 生物-生化与分子生物学
CiteScore
5.10
自引率
3.20%
发文量
134
审稿时长
1 months
期刊介绍: Molecular Genetics and Genomics (MGG) publishes peer-reviewed articles covering all areas of genetics and genomics. Any approach to the study of genes and genomes is considered, be it experimental, theoretical or synthetic. MGG publishes research on all organisms that is of broad interest to those working in the fields of genetics, genomics, biology, medicine and biotechnology. The journal investigates a broad range of topics, including these from recent issues: mechanisms for extending longevity in a variety of organisms; screening of yeast metal homeostasis genes involved in mitochondrial functions; molecular mapping of cultivar-specific avirulence genes in the rice blast fungus and more.
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