一个保加利亚家族三代人COL11A1基因突变导致马歇尔-斯蒂克勒综合征

IF 0.5 4区 医学 Q4 GENETICS & HEREDITY
Balkan Journal of Medical Genetics Pub Date : 2021-07-27 eCollection Date: 2021-06-01 DOI:10.2478/bjmg-2021-0001
M Mladenova, T Todorov, L Grozdanova, V Mitev, A Todorova
{"title":"一个保加利亚家族三代人COL11A1基因突变导致马歇尔-斯蒂克勒综合征","authors":"M Mladenova,&nbsp;T Todorov,&nbsp;L Grozdanova,&nbsp;V Mitev,&nbsp;A Todorova","doi":"10.2478/bjmg-2021-0001","DOIUrl":null,"url":null,"abstract":"<p><p>Here we report the first familial case spread through at least three generations, genetically verified cases of Marshall-Stickler syndrome in Bulgaria. The proband, a 2-year-old girl, has craniofacial dysplasia, ocular hypertelorism, small saddle nose with a flat bridge and midface hypoplasia. The pedigree of the proband's family showed that her father has the same clinical manifestations of the disease. In addition, her father presented with a tall, thin stature and mild hearing loss, manifested with aging. The same dysmorphological symptoms were presented by the paternal grandfather. Both patients, the 2-year-old girl and her father, have been diagnosed to carry Marshall-Stickler syndrome. The <i>COL2A1</i> gene tested negative in the family. Based on the higher percentage of mutations in the <i>COL2A1</i> gene, we analyzed this gene as the first target in the family. The <i>COL2A1</i> gene tested negative, and we sequenced the gene further. A novel splice site mutation c.3474+1G>A was found in intron 44. This variant is related to the clinical presentation in the patient and her father. The c.3474+1G>A mutation results in altered splicing affects at the donor splice site of intron 44, which most probably gives a nonfunctional protein. The variant affects the major triple-helical domain that represents a mutation hot-spot for the gene.</p>","PeriodicalId":55403,"journal":{"name":"Balkan Journal of Medical Genetics","volume":null,"pages":null},"PeriodicalIF":0.5000,"publicationDate":"2021-07-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/f8/57/bjmg-24-095.PMC8366474.pdf","citationCount":"3","resultStr":"{\"title\":\"Novel Mutation in the <i>COL11A1</i> Gene Causing Marshall-Stickler Syndrome in Three Generations of a Bulgarian Family.\",\"authors\":\"M Mladenova,&nbsp;T Todorov,&nbsp;L Grozdanova,&nbsp;V Mitev,&nbsp;A Todorova\",\"doi\":\"10.2478/bjmg-2021-0001\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Here we report the first familial case spread through at least three generations, genetically verified cases of Marshall-Stickler syndrome in Bulgaria. The proband, a 2-year-old girl, has craniofacial dysplasia, ocular hypertelorism, small saddle nose with a flat bridge and midface hypoplasia. The pedigree of the proband's family showed that her father has the same clinical manifestations of the disease. In addition, her father presented with a tall, thin stature and mild hearing loss, manifested with aging. The same dysmorphological symptoms were presented by the paternal grandfather. Both patients, the 2-year-old girl and her father, have been diagnosed to carry Marshall-Stickler syndrome. The <i>COL2A1</i> gene tested negative in the family. Based on the higher percentage of mutations in the <i>COL2A1</i> gene, we analyzed this gene as the first target in the family. The <i>COL2A1</i> gene tested negative, and we sequenced the gene further. A novel splice site mutation c.3474+1G>A was found in intron 44. This variant is related to the clinical presentation in the patient and her father. The c.3474+1G>A mutation results in altered splicing affects at the donor splice site of intron 44, which most probably gives a nonfunctional protein. The variant affects the major triple-helical domain that represents a mutation hot-spot for the gene.</p>\",\"PeriodicalId\":55403,\"journal\":{\"name\":\"Balkan Journal of Medical Genetics\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":0.5000,\"publicationDate\":\"2021-07-27\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/f8/57/bjmg-24-095.PMC8366474.pdf\",\"citationCount\":\"3\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Balkan Journal of Medical Genetics\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.2478/bjmg-2021-0001\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2021/6/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q4\",\"JCRName\":\"GENETICS & HEREDITY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Balkan Journal of Medical Genetics","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.2478/bjmg-2021-0001","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2021/6/1 0:00:00","PubModel":"eCollection","JCR":"Q4","JCRName":"GENETICS & HEREDITY","Score":null,"Total":0}
引用次数: 3

摘要

在这里,我们报告了第一个家族性病例传播至少三代,遗传验证的病例马歇尔-斯蒂克勒综合征在保加利亚。先证者为2岁女童,颅面发育不良,眼远视,鞍鼻小,鼻梁扁平,中脸发育不全。先证者的家谱显示她的父亲有同样的临床表现。此外,她的父亲身材瘦高,轻度听力下降,表现为衰老。祖父也表现出相同的畸形症状。两岁的女孩和她的父亲都被诊断患有马歇尔-斯蒂克勒综合征。该家族的COL2A1基因检测呈阴性。基于COL2A1基因较高的突变百分比,我们分析了该基因作为该家族的第一个目标。COL2A1基因检测呈阴性,我们对该基因进行了进一步测序。在内含子44中发现了一个新的剪接位点突变c.3474+1G>A。这种变异与患者及其父亲的临床表现有关。c.3474+1G>A突变导致内含子44供体剪接位点剪接作用发生改变,很可能产生无功能蛋白。这种变异影响了代表基因突变热点的主要三螺旋结构域。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Novel Mutation in the <i>COL11A1</i> Gene Causing Marshall-Stickler Syndrome in Three Generations of a Bulgarian Family.

Novel Mutation in the <i>COL11A1</i> Gene Causing Marshall-Stickler Syndrome in Three Generations of a Bulgarian Family.

Novel Mutation in the <i>COL11A1</i> Gene Causing Marshall-Stickler Syndrome in Three Generations of a Bulgarian Family.

Novel Mutation in the COL11A1 Gene Causing Marshall-Stickler Syndrome in Three Generations of a Bulgarian Family.

Here we report the first familial case spread through at least three generations, genetically verified cases of Marshall-Stickler syndrome in Bulgaria. The proband, a 2-year-old girl, has craniofacial dysplasia, ocular hypertelorism, small saddle nose with a flat bridge and midface hypoplasia. The pedigree of the proband's family showed that her father has the same clinical manifestations of the disease. In addition, her father presented with a tall, thin stature and mild hearing loss, manifested with aging. The same dysmorphological symptoms were presented by the paternal grandfather. Both patients, the 2-year-old girl and her father, have been diagnosed to carry Marshall-Stickler syndrome. The COL2A1 gene tested negative in the family. Based on the higher percentage of mutations in the COL2A1 gene, we analyzed this gene as the first target in the family. The COL2A1 gene tested negative, and we sequenced the gene further. A novel splice site mutation c.3474+1G>A was found in intron 44. This variant is related to the clinical presentation in the patient and her father. The c.3474+1G>A mutation results in altered splicing affects at the donor splice site of intron 44, which most probably gives a nonfunctional protein. The variant affects the major triple-helical domain that represents a mutation hot-spot for the gene.

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
CiteScore
1.00
自引率
0.00%
发文量
0
审稿时长
>12 weeks
期刊介绍: Balkan Journal of Medical Genetics is a journal in the English language for publication of articles involving all branches of medical genetics: human cytogenetics, molecular genetics, clinical genetics, immunogenetics, oncogenetics, pharmacogenetics, population genetics, genetic screening and diagnosis of monogenic and polygenic diseases, prenatal and preimplantation genetic diagnosis, genetic counselling, advances in treatment and prevention.
×
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:481959085
Book学术官方微信