与早发性马凡氏综合征严重表型相关的FBN1错义变异

IF 0.6 Q4 PEDIATRICS PROGRESS IN PEDIATRIC CARDIOLOGY Pub Date : 2024-08-28 DOI:10.1016/j.ppedcard.2024.101751
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
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引用次数: 0

摘要

早发性马凡综合征是马凡综合征(MFS)的一种罕见亚型,表现为生命早期发病。受影响的个体通常表现为严重的表型,早在新生儿期或婴幼儿期就出现关键的体征和症状,最常见的原因是新生致病性 FBN1 变异。我们描述了一名患有 FBN1 第 27 号外显子 c.3284G>C(p.(Cys1095Ser))罕见从头错义变异的患者,该变异可导致早发性 MFS,并伴有严重的心血管受累,包括心脏增大伴双侧房室瓣反流、主动脉扩张和反流,患者从出生起就表现出这些症状,最终死亡。
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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.

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来源期刊
CiteScore
0.90
自引率
11.10%
发文量
69
审稿时长
75 days
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.
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