Literature DB >> 20129799

Congenital stridor and wheezing as harbingers of the del22q11.2 syndrome presenting cardiovascular malformations of right aortic arch, aberrant left subclavian artery, Kommerell's diverticulum, and left ligamentum arteriosum.

Meng-Luen Lee1, Ming Chen, Lon-Yen Tsao, Han-Yao Chiu, Ing-Sh Chiu, Albert D Yang, Pei-Ling Tsai.   

Abstract

A complete vascular ring composed of right aortic arch, aberrant left subclavian artery with Kommerell's diverticulum, and left ligamentum arteriosum was diagnosed by barium esophagography, echocardiography, angiography, and multidetector computed tomography of chest in an 18-day-old male neonate who presented with remarkable inspiratory stridor, expiratory wheezing, postprandial vomiting, and dysphagia since birth, and survived surgical division of the left ligamentum arteriosum, resection of the Kommerell's diverticulum, and reimplanation of the left subclavian artery to the left common carotid artery. Cytogenetic analysis and fluorescence in situ hybridization study of his blood revealed chromosome 22q11.2 deletion, with a karyotype of 46,XY.ish del(22)(q11.2 q11.2). A constellation of right aortic arch, aberrant left subclavian artery with Kommerell's diverticulum, and left ligamentum arteriosum in neonates may cause refractory stridor, wheezing, vomiting, and dysphagia, which can serve as harbingers of the del22q11.2 syndrome.
Copyright © 2011 Elsevier Inc. All rights reserved.

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Year:  2010        PMID: 20129799     DOI: 10.1016/j.carpath.2010.01.006

Source DB:  PubMed          Journal:  Cardiovasc Pathol        ISSN: 1054-8807            Impact factor:   2.185


  1 in total

1.  Combined interventional and surgical treatment for a rare case of double patent ductus arteriosus.

Authors:  Xiao-Ke Shang; Gang-Cheng Zhang; Liang Zhong; Xin Zhou; Mei Liu; Rong Lu
Journal:  Exp Ther Med       Date:  2015-12-08       Impact factor: 2.447

  1 in total

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