Sara Markholt , Jens Skaerbaek , Kim Munk , Brian N. Andersen , Dorte L. Lilballe , Jenny Blechingberg , Jesper P. Petersen , Jesper V. Bjerre , Pernille A. Gregersen , Kasper J. Kyng
{"title":"与早发性马凡氏综合征严重表型相关的FBN1错义变异","authors":"Sara Markholt , Jens Skaerbaek , Kim Munk , Brian N. Andersen , Dorte L. Lilballe , Jenny Blechingberg , Jesper P. Petersen , Jesper V. Bjerre , Pernille A. Gregersen , Kasper J. Kyng","doi":"10.1016/j.ppedcard.2024.101751","DOIUrl":null,"url":null,"abstract":"<div><p>Early-onset Marfan syndrome is a rare subtype of Marfan Syndrome (MFS), manifesting early in life. Affected individuals typically present with a severe phenotype, with critical signs and symptoms as early as the neonatal or infantile period, most often due to a <em>de novo</em> pathogenic <em>FBN1</em> variant. We describe a patient with a rare <em>de novo</em> missense variant c.3284G>C (p.(Cys1095Ser)) in exon 27 in <em>FBN1</em> leading to early onset MFS with severe cardiovascular involvement including heart enlargement with regurgitation of both atrioventricular valves, aortic dilatation and regurgitation manifest from birth, with a fatal outcome.</p></div>","PeriodicalId":46028,"journal":{"name":"PROGRESS IN PEDIATRIC CARDIOLOGY","volume":null,"pages":null},"PeriodicalIF":0.6000,"publicationDate":"2024-08-28","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S1058981324000493/pdfft?md5=e26303f111c9d5b310bd45292b186d98&pid=1-s2.0-S1058981324000493-main.pdf","citationCount":"0","resultStr":"{\"title\":\"A de novo FBN1 missense variant associated with a severe phenotype of early onset Marfan syndrome\",\"authors\":\"Sara Markholt , Jens Skaerbaek , Kim Munk , Brian N. Andersen , Dorte L. Lilballe , Jenny Blechingberg , Jesper P. Petersen , Jesper V. Bjerre , Pernille A. Gregersen , Kasper J. Kyng\",\"doi\":\"10.1016/j.ppedcard.2024.101751\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><p>Early-onset Marfan syndrome is a rare subtype of Marfan Syndrome (MFS), manifesting early in life. Affected individuals typically present with a severe phenotype, with critical signs and symptoms as early as the neonatal or infantile period, most often due to a <em>de novo</em> pathogenic <em>FBN1</em> variant. We describe a patient with a rare <em>de novo</em> missense variant c.3284G>C (p.(Cys1095Ser)) in exon 27 in <em>FBN1</em> leading to early onset MFS with severe cardiovascular involvement including heart enlargement with regurgitation of both atrioventricular valves, aortic dilatation and regurgitation manifest from birth, with a fatal outcome.</p></div>\",\"PeriodicalId\":46028,\"journal\":{\"name\":\"PROGRESS IN PEDIATRIC CARDIOLOGY\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":0.6000,\"publicationDate\":\"2024-08-28\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.sciencedirect.com/science/article/pii/S1058981324000493/pdfft?md5=e26303f111c9d5b310bd45292b186d98&pid=1-s2.0-S1058981324000493-main.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"PROGRESS IN PEDIATRIC CARDIOLOGY\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S1058981324000493\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"PEDIATRICS\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"PROGRESS IN PEDIATRIC CARDIOLOGY","FirstCategoryId":"1085","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S1058981324000493","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"PEDIATRICS","Score":null,"Total":0}
A de novo FBN1 missense variant associated with a severe phenotype of early onset Marfan syndrome
Early-onset Marfan syndrome is a rare subtype of Marfan Syndrome (MFS), manifesting early in life. Affected individuals typically present with a severe phenotype, with critical signs and symptoms as early as the neonatal or infantile period, most often due to a de novo pathogenic FBN1 variant. We describe a patient with a rare de novo missense variant c.3284G>C (p.(Cys1095Ser)) in exon 27 in FBN1 leading to early onset MFS with severe cardiovascular involvement including heart enlargement with regurgitation of both atrioventricular valves, aortic dilatation and regurgitation manifest from birth, with a fatal outcome.
期刊介绍:
Progress in Pediatric Cardiology is an international journal of review presenting information and experienced opinion of importance in the understanding and management of cardiovascular diseases in children. Each issue is prepared by one or more Guest Editors and reviews a single subject, allowing for comprehensive presentations of complex, multifaceted or rapidly changing topics of clinical and investigative interest.