De Novo interstitial deletion 13q33.3q34 in a male patient with double outlet right ventricle, microcephaly, dysmorphic craniofacial findings, and motor and developmental delay

Am J Med Genet A. 2015 May;167A(5):1134-41. doi: 10.1002/ajmg.a.36978. Epub 2015 Mar 21.

Abstract

We describe a 6-year-old male, diagnosed at birth with double outlet right ventricle (DORV), anterior aorta, multiple ventricular septal defects, pulmonary stenosis, microcephaly and mildly dysmorphic craniofacial findings. Chromosomal analysis showed a normal male karyotype but on subsequent array comparative genomic hybridization (array CGH) analysis a de novo 2.5 Mb loss in chromosome 13q at 13q33.3q34, together with an inherited gain at 4p12, were detected. The propositus underwent placement of a Blalock Taussig shunt and subsequently a Glenn and Fontan operation was performed. In this report we propose that COL4A1 and COL4A2 may be candidate genes for congenital heart disease (CHD) in individuals with a deletion in 13q within the 6Mb critical region for cardiac development proposed by Huang et al., [2012].

Keywords: COL4A1; COL4A2; CORIN; Congenital heart defect (CHD); array comparative genomic hybridization (array CGH); chromosome 13q33.3q34 deletion; double outlet right ventricle (DORV); dysmorphic craniofacial findings; inherited paternal gain at 4p12.

Publication types

  • Case Reports

MeSH terms

  • Child
  • Chromosome Deletion*
  • Chromosomes, Human, Pair 13 / genetics
  • Collagen Type IV / genetics
  • Comparative Genomic Hybridization
  • Craniofacial Abnormalities / genetics*
  • Craniofacial Abnormalities / physiopathology
  • Developmental Disabilities / genetics
  • Developmental Disabilities / physiopathology
  • Double Outlet Right Ventricle / genetics*
  • Double Outlet Right Ventricle / physiopathology
  • Genetic Association Studies
  • Heart Defects, Congenital / genetics*
  • Heart Defects, Congenital / physiopathology
  • Humans
  • Male
  • Microcephaly / genetics*
  • Microcephaly / physiopathology
  • Serine Endopeptidases / genetics

Substances

  • COL4A1 protein, human
  • COL4A2 protein, human
  • Collagen Type IV
  • CORIN protein, human
  • Serine Endopeptidases