Literature DB >> 25428557

Aortopathy in the 7q11.23 microduplication syndrome.

Ashley Parrott1, Jeanne James, Paula Goldenberg, Robert B Hinton, Erin Miller, Amy Shikany, Arthur S Aylsworth, Kathleen Kaiser-Rogers, Sunita J Ferns, Seema R Lalani, Stephanie M Ware.   

Abstract

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.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  7q11.23 microduplication; Williams-Beuren syndrome; elastin; pediatrics; thoracic aortic aneurysm

Mesh:

Year:  2014        PMID: 25428557     DOI: 10.1002/ajmg.a.36859

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  10 in total

1.  Children with 7q11.23 duplication syndrome: psychological characteristics.

Authors:  Carolyn B Mervis; Bonita P Klein-Tasman; Myra J Huffman; Shelley L Velleman; C Holley Pitts; Danielle R Henderson; Janet Woodruff-Borden; Colleen A Morris; Lucy R Osborne
Journal:  Am J Med Genet A       Date:  2015-04-21       Impact factor: 2.802

2.  7q11.23 Duplication syndrome: Physical characteristics and natural history.

Authors:  Colleen A Morris; Carolyn B Mervis; Alex P Paciorkowski; Omar Abdul-Rahman; Sarah L Dugan; Alan F Rope; Patricia Bader; Laura G Hendon; Shelley L Velleman; Bonita P Klein-Tasman; Lucy R Osborne
Journal:  Am J Med Genet A       Date:  2015-09-03       Impact factor: 2.802

Review 3.  The 7q11.23 Microduplication Syndrome: A Clinical Report with Review of Literature.

Authors:  Elham Abbas; Devin M Cox; Teri Smith; Merlin G Butler
Journal:  J Pediatr Genet       Date:  2016-06-15

Review 4.  Cytogenomic Aberrations in Congenital Cardiovascular Malformations.

Authors:  Mahshid Azamian; Seema R Lalani
Journal:  Mol Syndromol       Date:  2016-04-26

5.  Aortic dilation, genetic testing, and associated diagnoses.

Authors:  Yuri A Zarate; Elizabeth Sellars; Tiffany Lepard; Xinyu Tang; R Thomas Collins
Journal:  Genet Med       Date:  2015-07-02       Impact factor: 8.822

6.  Computerized Tomography Use in Williams-Beuren Syndrome Aortopathy.

Authors:  Neale Nicola Kalis; Leena Khalifa Sulaibikh; Saud Rashid Al Amer; Haya Yousif Al Amer
Journal:  Heart Views       Date:  2017 Jan-Mar

7.  A Novel Human Biospecimen Repository for Clinical and Molecular Investigation of Thoracic Aortopathy.

Authors:  Courtney E Vujakovich; Benjamin J Landis
Journal:  Cardiogenetics       Date:  2021-09-18

8.  Rationale for the Cytogenomics of Cardiovascular Malformations Consortium: A Phenotype Intensive Registry Based Approach.

Authors:  Robert B Hinton; Kim L McBride; Steven B Bleyl; Neil E Bowles; William L Border; Vidu Garg; Teresa A Smolarek; Seema R Lalani; Stephanie M Ware
Journal:  J Cardiovasc Dev Dis       Date:  2015-04-29

9.  A method for determining haploid and triploid genotypes and their association with vascular phenotypes in Williams syndrome and 7q11.23 duplication syndrome.

Authors:  Michael D Gregory; Bhaskar Kolachana; Yin Yao; Tiffany Nash; Dwight Dickinson; Daniel P Eisenberg; Carolyn B Mervis; Karen F Berman
Journal:  BMC Med Genet       Date:  2018-04-04       Impact factor: 2.103

10.  Copy number variations associated with fetal congenital kidney malformations.

Authors:  Meiying Cai; Na Lin; Linjuan Su; Xiaoqing Wu; Xiaorui Xie; Ying Li; Xuemei Chen; Yuan Lin; Hailong Huang; Liangpu Xu
Journal:  Mol Cytogenet       Date:  2020-03-24       Impact factor: 2.009

  10 in total

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