Sarah Østergaard Holm, Lise Graversen, Anne Katrine Wrist Simonsen, Jenny Blechingberg, Laura Krogh Herlin, Kirsten Rønholt
{"title":"一个被诊断为由HRURF新变异引起的Marie Unna遗传性毛少症1的家庭的临床表现。","authors":"Sarah Østergaard Holm, Lise Graversen, Anne Katrine Wrist Simonsen, Jenny Blechingberg, Laura Krogh Herlin, Kirsten Rønholt","doi":"10.1111/pde.15982","DOIUrl":null,"url":null,"abstract":"<p><p>Marie Unna hereditary hypotrichosis 1 (MUHH1) is a rare autosomal dominant condition characterized by the absence or scarcity of hair at birth with the growth of coarse, wiry, and unruly hair during childhood and progressive hair loss after puberty. This case report describes the clinical presentation of three Danish family members with congenital hypotrichosis with a new phenotypic variation marked by progressive scalp hair growth during adulthood in one of the members. A novel likely pathogenic variant c.105G>T (p.(*35Tyrext*?)) in HRURF, formerly known as U2HR, causing MUHH1 was identified. The findings contribute to the knowledge on pathogenic and phenotypic variation in MUHH1.</p>","PeriodicalId":19819,"journal":{"name":"Pediatric Dermatology","volume":" ","pages":""},"PeriodicalIF":1.2000,"publicationDate":"2025-05-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Clinical Presentation of a Family Diagnosed With Marie Unna Hereditary Hypotrichosis 1 Caused by a Novel Variant in HRURF.\",\"authors\":\"Sarah Østergaard Holm, Lise Graversen, Anne Katrine Wrist Simonsen, Jenny Blechingberg, Laura Krogh Herlin, Kirsten Rønholt\",\"doi\":\"10.1111/pde.15982\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Marie Unna hereditary hypotrichosis 1 (MUHH1) is a rare autosomal dominant condition characterized by the absence or scarcity of hair at birth with the growth of coarse, wiry, and unruly hair during childhood and progressive hair loss after puberty. This case report describes the clinical presentation of three Danish family members with congenital hypotrichosis with a new phenotypic variation marked by progressive scalp hair growth during adulthood in one of the members. A novel likely pathogenic variant c.105G>T (p.(*35Tyrext*?)) in HRURF, formerly known as U2HR, causing MUHH1 was identified. The findings contribute to the knowledge on pathogenic and phenotypic variation in MUHH1.</p>\",\"PeriodicalId\":19819,\"journal\":{\"name\":\"Pediatric Dermatology\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":1.2000,\"publicationDate\":\"2025-05-28\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pediatric Dermatology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1111/pde.15982\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"DERMATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pediatric Dermatology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1111/pde.15982","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"DERMATOLOGY","Score":null,"Total":0}
引用次数: 0
摘要
Marie Unna遗传性毛少症1 (muh1)是一种罕见的常染色体显性遗传病,其特征是出生时没有或缺乏头发,儿童时期生长粗糙,坚硬和不守规矩的头发,青春期后逐渐脱发。本病例报告描述了三个丹麦家庭成员先天性毛少症的临床表现,其中一个家庭成员在成年期出现了一种新的表型变异,其特征是头皮毛发逐渐生长。在HRURF(以前称为U2HR)中发现了一种新的可能致病变异c.105G >t (p.(* 35tyext *?)),导致muh1。这些发现有助于了解muh1的致病和表型变异。
Clinical Presentation of a Family Diagnosed With Marie Unna Hereditary Hypotrichosis 1 Caused by a Novel Variant in HRURF.
Marie Unna hereditary hypotrichosis 1 (MUHH1) is a rare autosomal dominant condition characterized by the absence or scarcity of hair at birth with the growth of coarse, wiry, and unruly hair during childhood and progressive hair loss after puberty. This case report describes the clinical presentation of three Danish family members with congenital hypotrichosis with a new phenotypic variation marked by progressive scalp hair growth during adulthood in one of the members. A novel likely pathogenic variant c.105G>T (p.(*35Tyrext*?)) in HRURF, formerly known as U2HR, causing MUHH1 was identified. The findings contribute to the knowledge on pathogenic and phenotypic variation in MUHH1.
期刊介绍:
Pediatric Dermatology answers the need for new ideas and strategies for today''s pediatrician or dermatologist. As a teaching vehicle, the Journal is still unsurpassed and it will continue to present the latest on topics such as hemangiomas, atopic dermatitis, rare and unusual presentations of childhood diseases, neonatal medicine, and therapeutic advances. As important progress is made in any area involving infants and children, Pediatric Dermatology is there to publish the findings.