在一个由 10 个家族组成的队列中,HR 中的新型致病变体是富贵病伴丘疹性病变的基础。

Kifayat Ullah, Sohail Ahmed, Nicole Cesarato, Xing Xiong, Maria Taj, Naushaba Manan, Maria Wehner, Muhammad Javed Khan, Hammal Khan, Sabba Mehmood, Muhammad Sharif Hasni, Dariusz Michna, Rehmana Waris, Henning Hamm, Regina C Betz, Wasim Ahmad, Imran Ullah
{"title":"在一个由 10 个家族组成的队列中,HR 中的新型致病变体是富贵病伴丘疹性病变的基础。","authors":"Kifayat Ullah, Sohail Ahmed, Nicole Cesarato, Xing Xiong, Maria Taj, Naushaba Manan, Maria Wehner, Muhammad Javed Khan, Hammal Khan, Sabba Mehmood, Muhammad Sharif Hasni, Dariusz Michna, Rehmana Waris, Henning Hamm, Regina C Betz, Wasim Ahmad, Imran Ullah","doi":"10.1111/1346-8138.17349","DOIUrl":null,"url":null,"abstract":"<p><p>Atrichia with papular lesions (APL) is a hair abnormality characterized by loss of hair on the scalp and rest of the body. In a few cases, hair loss is accompanied by the appearance of keratotic papules on the body. It is inherited in an autosomal recessive manner. Sequence variants in the HR (hairless) gene are responsible for this hair abnormality. Here, we present nine consanguineous families and one nonconsanguineous family with clinical manifestations of APL. Whole exome followed by Sanger sequencing and/or direct Sanger sequencing was performed to identify pathogenic variants. The study revealed seven novel pathogenic variants c.794del;p.(Pro265Argfs*98), c.2921-2936del;p.(Tyr974Leufs*16), c.2889C>A;p.(Cys963*), c.2689C>T;p.(Gln897*), c.3186_3187dup;p.(Gln1063Profs*43), c.560dup;p.(Tyr188Ilefs*131), c.2203+5G>C, c.2776+5G>A, and the previously reported variant c.1837C>T;p.(Arg613*) in HR in these families. The study not only expands the mutational spectrum in the HR gene but also highlights the unusual phenotypic findings and will facilitate genetic counseling of families with members showing various types of hair loss disorders in the local population.</p>","PeriodicalId":94236,"journal":{"name":"The Journal of dermatology","volume":null,"pages":null},"PeriodicalIF":0.0000,"publicationDate":"2024-06-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Novel pathogenic variants in HR underlie atrichia with papular lesions in a cohort of 10 families.\",\"authors\":\"Kifayat Ullah, Sohail Ahmed, Nicole Cesarato, Xing Xiong, Maria Taj, Naushaba Manan, Maria Wehner, Muhammad Javed Khan, Hammal Khan, Sabba Mehmood, Muhammad Sharif Hasni, Dariusz Michna, Rehmana Waris, Henning Hamm, Regina C Betz, Wasim Ahmad, Imran Ullah\",\"doi\":\"10.1111/1346-8138.17349\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Atrichia with papular lesions (APL) is a hair abnormality characterized by loss of hair on the scalp and rest of the body. In a few cases, hair loss is accompanied by the appearance of keratotic papules on the body. It is inherited in an autosomal recessive manner. Sequence variants in the HR (hairless) gene are responsible for this hair abnormality. Here, we present nine consanguineous families and one nonconsanguineous family with clinical manifestations of APL. Whole exome followed by Sanger sequencing and/or direct Sanger sequencing was performed to identify pathogenic variants. The study revealed seven novel pathogenic variants c.794del;p.(Pro265Argfs*98), c.2921-2936del;p.(Tyr974Leufs*16), c.2889C>A;p.(Cys963*), c.2689C>T;p.(Gln897*), c.3186_3187dup;p.(Gln1063Profs*43), c.560dup;p.(Tyr188Ilefs*131), c.2203+5G>C, c.2776+5G>A, and the previously reported variant c.1837C>T;p.(Arg613*) in HR in these families. The study not only expands the mutational spectrum in the HR gene but also highlights the unusual phenotypic findings and will facilitate genetic counseling of families with members showing various types of hair loss disorders in the local population.</p>\",\"PeriodicalId\":94236,\"journal\":{\"name\":\"The Journal of dermatology\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2024-06-24\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"The Journal of dermatology\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1111/1346-8138.17349\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"The Journal of dermatology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1111/1346-8138.17349","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0

摘要

毛囊角化性丘疹病(APL)是一种毛发异常,其特征是头皮和身体其他部位的毛发脱落。在少数病例中,脱发会伴随身体出现角化性丘疹。该病为常染色体隐性遗传。HR(无毛)基因的序列变异是导致这种毛发异常的原因。在此,我们介绍了具有 APL 临床表现的九个近亲家族和一个非近亲家族。为了确定致病变体,我们进行了全外显子组测序和/或直接桑格测序。研究发现了 7 个新的致病变体:c.794del;p. (Pro265Argfs*98)、c.2921-2936del;p. (Tyr974Leufs*16)、c.2889C>A;p. (Cys963*)、c.2689C>T;p. (Gln897*)、c.3186_3187dup;p.(Gln1063Profs*43)、c.560dup;p.(Tyr188Ilefs*131)、c.2203+5G>C、c.2776+5G>A,以及之前报道的这些家族中 HR 变异 c.1837C>T;p.(Arg613*)。这项研究不仅扩大了 HR 基因的突变范围,而且突出了不寻常的表型发现,将有助于为本地人群中出现各种脱发疾病的家族成员提供遗传咨询。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
Novel pathogenic variants in HR underlie atrichia with papular lesions in a cohort of 10 families.

Atrichia with papular lesions (APL) is a hair abnormality characterized by loss of hair on the scalp and rest of the body. In a few cases, hair loss is accompanied by the appearance of keratotic papules on the body. It is inherited in an autosomal recessive manner. Sequence variants in the HR (hairless) gene are responsible for this hair abnormality. Here, we present nine consanguineous families and one nonconsanguineous family with clinical manifestations of APL. Whole exome followed by Sanger sequencing and/or direct Sanger sequencing was performed to identify pathogenic variants. The study revealed seven novel pathogenic variants c.794del;p.(Pro265Argfs*98), c.2921-2936del;p.(Tyr974Leufs*16), c.2889C>A;p.(Cys963*), c.2689C>T;p.(Gln897*), c.3186_3187dup;p.(Gln1063Profs*43), c.560dup;p.(Tyr188Ilefs*131), c.2203+5G>C, c.2776+5G>A, and the previously reported variant c.1837C>T;p.(Arg613*) in HR in these families. The study not only expands the mutational spectrum in the HR gene but also highlights the unusual phenotypic findings and will facilitate genetic counseling of families with members showing various types of hair loss disorders in the local 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
自引率
0.00%
发文量
0
期刊最新文献
Clinical and laboratory features between anti‐TIF1γ dermatomyositis with and without malignancy: 37 case series and a review Analysis of disease burden in patients with hereditary angioedema from Japan by patient‐reported outcomes Coexistence of Basan syndrome and cutaneous basal cell carcinoma: Genetic and clinical perspectives Perspectives of Japanese patients on psoriatic disease burden: Results from “Psoriasis and Beyond,” the Global Psoriatic Disease Survey HPV 51‐associated inguinal SCC on an atopic dermatitis patient treated with cyclosporine
×
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
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 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 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1