土耳其肌糖病患者SGCG的一个创始等位基因,结合错义变异和多外显子重复。

IF 2.3 3区 医学 Q2 GENETICS & HEREDITY
Abdullah Sezer, Afife Büke, Hasan Hüseyin Kazan, Ahmet Kablan, Mustafa Tarık Alay, Beril Talim, Deniz Yüksel, Hanife Saat
{"title":"土耳其肌糖病患者SGCG的一个创始等位基因,结合错义变异和多外显子重复。","authors":"Abdullah Sezer, Afife Büke, Hasan Hüseyin Kazan, Ahmet Kablan, Mustafa Tarık Alay, Beril Talim, Deniz Yüksel, Hanife Saat","doi":"10.1111/cge.70041","DOIUrl":null,"url":null,"abstract":"<p><p>Sarcoglycanopathies are autosomal recessive muscular dystrophies characterized by progressive muscle weakness and represent a major subset of limb-girdle muscular dystrophies (LGMDs). They result from pathogenic variants in sarcoglycan genes (SGCA, SGCB, SGCD, and SGCG), which encode subunits of a tetrameric transmembrane complex that stabilizes the dystrophin-associated glycoprotein complex. Among these, SGCG is commonly affected and is associated with LGMD2C/R5. Several founder variants in SGCG have been reported across different populations. Here, we describe a novel founder allele in the Turkish population, comprising a missense variant (c.392A>G, p.Lys131Arg) and a copy number gain (exon 1-4 duplication) that occurs in cis. Through an inductive screening strategy, we identified these linked variants in 11 individuals: five affected patients from four families and six unrelated incidental carriers. Haplotype analysis confirmed a shared genomic background supporting a founder effect. Affected individuals carried the allele in a homozygous or compound heterozygous state with other pathogenic SGCG variants. Molecular and histopathological investigations revealed that the duplicated allele results in mRNA decay and loss of SGCG protein expression. This study represents the first report of a disease-associated duplication in SGCG, highlighting a novel founder allele composed of two linked variants that contribute to sarcoglycanopathy in a specific population.</p>","PeriodicalId":10354,"journal":{"name":"Clinical Genetics","volume":" ","pages":""},"PeriodicalIF":2.3000,"publicationDate":"2025-08-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"A Founder Allele in SGCG Combining Missense Variant and Multi-Exon Duplication in Turkish Patients With Sarcoglycanopathy.\",\"authors\":\"Abdullah Sezer, Afife Büke, Hasan Hüseyin Kazan, Ahmet Kablan, Mustafa Tarık Alay, Beril Talim, Deniz Yüksel, Hanife Saat\",\"doi\":\"10.1111/cge.70041\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Sarcoglycanopathies are autosomal recessive muscular dystrophies characterized by progressive muscle weakness and represent a major subset of limb-girdle muscular dystrophies (LGMDs). They result from pathogenic variants in sarcoglycan genes (SGCA, SGCB, SGCD, and SGCG), which encode subunits of a tetrameric transmembrane complex that stabilizes the dystrophin-associated glycoprotein complex. Among these, SGCG is commonly affected and is associated with LGMD2C/R5. Several founder variants in SGCG have been reported across different populations. Here, we describe a novel founder allele in the Turkish population, comprising a missense variant (c.392A>G, p.Lys131Arg) and a copy number gain (exon 1-4 duplication) that occurs in cis. Through an inductive screening strategy, we identified these linked variants in 11 individuals: five affected patients from four families and six unrelated incidental carriers. Haplotype analysis confirmed a shared genomic background supporting a founder effect. Affected individuals carried the allele in a homozygous or compound heterozygous state with other pathogenic SGCG variants. Molecular and histopathological investigations revealed that the duplicated allele results in mRNA decay and loss of SGCG protein expression. This study represents the first report of a disease-associated duplication in SGCG, highlighting a novel founder allele composed of two linked variants that contribute to sarcoglycanopathy in a specific population.</p>\",\"PeriodicalId\":10354,\"journal\":{\"name\":\"Clinical Genetics\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":2.3000,\"publicationDate\":\"2025-08-10\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Clinical Genetics\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1111/cge.70041\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"GENETICS & HEREDITY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Clinical Genetics","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1111/cge.70041","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"GENETICS & HEREDITY","Score":null,"Total":0}
引用次数: 0

摘要

肌糖病是常染色体隐性肌营养不良症,以进行性肌无力为特征,是肢带性肌营养不良症(LGMDs)的一个主要亚型。它们是由肌糖聚糖基因(SGCA、SGCB、SGCD和SGCG)的致病变异引起的,这些基因编码四聚体跨膜复合物的亚基,稳定肌营养不良蛋白相关的糖蛋白复合物。其中,SGCG常受影响,并与LGMD2C/R5相关。在不同的人群中已经报道了SGCG的几种创始变异。在这里,我们描述了土耳其人群中的一个新的创始等位基因,包括一个错义变体(c.392A>G, p.Lys131Arg)和一个拷贝数增加(外显子1-4重复),发生在顺式中。通过归纳筛选策略,我们在11个人中确定了这些相关变异:来自四个家庭的5名受影响患者和6名不相关的偶然携带者。单倍型分析证实了共同的基因组背景支持创始人效应。受影响个体携带的等位基因与其他致病性SGCG变异呈纯合或复合杂合状态。分子和组织病理学研究表明,重复的等位基因导致mRNA衰减和SGCG蛋白表达丧失。这项研究首次报道了SGCG中与疾病相关的重复,强调了一个由两个相关变异组成的新的创始等位基因,该等位基因在特定人群中导致肌糖病。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
A Founder Allele in SGCG Combining Missense Variant and Multi-Exon Duplication in Turkish Patients With Sarcoglycanopathy.

Sarcoglycanopathies are autosomal recessive muscular dystrophies characterized by progressive muscle weakness and represent a major subset of limb-girdle muscular dystrophies (LGMDs). They result from pathogenic variants in sarcoglycan genes (SGCA, SGCB, SGCD, and SGCG), which encode subunits of a tetrameric transmembrane complex that stabilizes the dystrophin-associated glycoprotein complex. Among these, SGCG is commonly affected and is associated with LGMD2C/R5. Several founder variants in SGCG have been reported across different populations. Here, we describe a novel founder allele in the Turkish population, comprising a missense variant (c.392A>G, p.Lys131Arg) and a copy number gain (exon 1-4 duplication) that occurs in cis. Through an inductive screening strategy, we identified these linked variants in 11 individuals: five affected patients from four families and six unrelated incidental carriers. Haplotype analysis confirmed a shared genomic background supporting a founder effect. Affected individuals carried the allele in a homozygous or compound heterozygous state with other pathogenic SGCG variants. Molecular and histopathological investigations revealed that the duplicated allele results in mRNA decay and loss of SGCG protein expression. This study represents the first report of a disease-associated duplication in SGCG, highlighting a novel founder allele composed of two linked variants that contribute to sarcoglycanopathy in a specific population.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
Clinical Genetics
Clinical Genetics 医学-遗传学
CiteScore
6.50
自引率
0.00%
发文量
175
审稿时长
3-8 weeks
期刊介绍: Clinical Genetics links research to the clinic, translating advances in our understanding of the molecular basis of genetic disease for the practising clinical geneticist. The journal publishes high quality research papers, short reports, reviews and mini-reviews that connect medical genetics research with clinical practice. Topics of particular interest are: • Linking genetic variations to disease • Genome rearrangements and disease • Epigenetics and disease • The translation of genotype to phenotype • Genetics of complex disease • Management/intervention of genetic diseases • Novel therapies for genetic diseases • Developmental biology, as it relates to clinical genetics • Social science research on the psychological and behavioural aspects of living with or being at risk of genetic disease
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:604180095
Book学术官方微信