Heterotaxy and cardiac defect in a girl with chromosome translocation t(X;1)(q26;p13.1) and involvement of ZIC3

Eur J Hum Genet. 2006 Dec;14(12):1317-20. doi: 10.1038/sj.ejhg.5201707. Epub 2006 Aug 23.

Abstract

We report on a 2-year-old girl with situs ambiguus comprising right-sided stomach and spleen, left-sided liver and complex cardiac defect. Psychomotor development of this patient was normal, and no other major abnormalities were present. Chromosome analysis revealed a de novo balanced chromosome translocation t(X;1)(q26;p13.1). Molecular cytogenetic investigations identified a breakpoint spanning BAC clone on the X-chromosome containing the ZIC3 gene. Mutations in ZIC3 are associated with situs ambiguus and cardiac defects predominantly in males. This is the first report of a live born girl with an X-autosome translocation involving the ZIC3 region.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Child, Preschool
  • Chromosomes, Human, Pair 1*
  • Chromosomes, Human, X*
  • Female
  • Fetal Heart / abnormalities*
  • Fetal Heart / diagnostic imaging
  • Heart Diseases / genetics*
  • Homeodomain Proteins / genetics*
  • Humans
  • Radiography
  • Transcription Factors / genetics*
  • Translocation, Genetic / genetics*

Substances

  • Homeodomain Proteins
  • Transcription Factors
  • ZIC3 protein, human