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PMID: 25428557 已发表 · ppublish 英语

Aortopathy in the 7q11.23 microduplication syndrome.

American journal of medical genetics. Part A ·第 167A 卷 ·第 2 期 ·2015-09-16

Parrott Ashley, James Jeanne, Goldenberg Paula, Hinton Robert B, Miller Erin, Shikany Amy, Aylsworth Arthur S, Kaiser-Rogers Kathleen, Ferns Sunita J, Lalani Seema R, Ware Stephanie M

摘要

The 7q11.23 microduplication syndrome, caused by the reciprocal duplication of the Williams-Beuren syndrome deletion region, is a genomic disorder with an emerging clinical phenotype. Dysmorphic features, congenital anomalies, hypotonia, developmental delay highlighted by variable speech delay, and autistic features are characteristic findings. Congenital heart defects, most commonly patent ductus arteriosus, have been reported in a minority of cases. Included in the duplicated region is elastin (ELN), implicated as the cause of supravalvar aortic stenosis in patients with Williams-Beuren syndrome. Here we present a series of eight pediatric patients and one adult with 7q11.23 microduplication syndrome, all of whom had aortic dilation, the opposite vascular phenotype of the typical supravalvar aortic stenosis found in Williams-Beuren syndrome. The ascending aorta was most commonly involved, while dilation was less frequently identified at the aortic root and sinotubular junction. The findings in these patients support a recommendation for cardiovascular surveillance in patients with 7q11.23 microduplication syndrome.

关键词
7q11.23 microduplication Williams-Beuren syndrome elastin pediatrics thoracic aortic aneurysm
文献信息
期刊
American journal of medical genetics. Part A
期刊简称
Am J Med Genet A
发表日期
2015-09-16
收录日期
2015-01-21
更新日期
2016-11-25
语言
英语
国家/地区
United States
NLM ID
101235741
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