Islam Uddin, Iqra Zafar, Caoling Xu, Wenqing Li, Muhammad Imran Khan, Limin Wu, Jianqiang Bao
{"title":"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。","authors":"Islam Uddin, Iqra Zafar, Caoling Xu, Wenqing Li, Muhammad Imran Khan, Limin Wu, Jianqiang Bao","doi":"10.1007/s00438-024-02205-7","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":18816,"journal":{"name":"Molecular Genetics and Genomics","volume":"299 1","pages":"111"},"PeriodicalIF":2.3000,"publicationDate":"2024-12-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Whole-exome sequencing identifies rare recessive variants in azoospermia patients from consanguineous Pakistani families.\",\"authors\":\"Islam Uddin, Iqra Zafar, Caoling Xu, Wenqing Li, Muhammad Imran Khan, Limin Wu, Jianqiang Bao\",\"doi\":\"10.1007/s00438-024-02205-7\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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.</p>\",\"PeriodicalId\":18816,\"journal\":{\"name\":\"Molecular Genetics and Genomics\",\"volume\":\"299 1\",\"pages\":\"111\"},\"PeriodicalIF\":2.3000,\"publicationDate\":\"2024-12-03\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Molecular Genetics and Genomics\",\"FirstCategoryId\":\"99\",\"ListUrlMain\":\"https://doi.org/10.1007/s00438-024-02205-7\",\"RegionNum\":3,\"RegionCategory\":\"生物学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"BIOCHEMISTRY & MOLECULAR BIOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Molecular Genetics and Genomics","FirstCategoryId":"99","ListUrlMain":"https://doi.org/10.1007/s00438-024-02205-7","RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"BIOCHEMISTRY & MOLECULAR BIOLOGY","Score":null,"Total":0}
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.
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.