22q12.3-q13.1 microdeletion including SOX10 causes atypical Waardenburg syndrome

Eur J Ophthalmol. 2021 Jul;31(4):2127-2134. doi: 10.1177/1120672120944350. Epub 2020 Jul 24.

Abstract

Purpose: To identify disease associated mutations in a male infant with congenital heart defects and heterochromia.

Methods: A detailed clinical examination and routine laboratory tests were performed on the patient. We applied whole exome sequencing to identify the causal mutation on the proband and other family members.

Results: The patient presented with severe congenital heart disease, strabismus, and pigment disturbances of the iris. We identified a deletion of 1.99 megabase [arr[hg19]22q12.3-13.1 (chr22:36656004-38643920) *1], including SOX10 and 13 RefSeq genes on this patient, which was associated with atypical Waardenburg syndrome.

Conclusion: Our results suggest that a deletion of 1.99 megabase (including SOX10) acts as a dominant pathogenic variant on the clinical presentations of this patient with atypical Waardenburg syndrome.

Keywords: Microdeletion; Waardenburg syndrome; heterochromia; next-generation sequencing.

Publication types

  • Case Reports

MeSH terms

  • Chromosomes, Human, Pair 22
  • Exome Sequencing
  • Humans
  • Infant
  • Iris
  • Male
  • Mutation
  • Pedigree
  • SOXE Transcription Factors / genetics
  • Waardenburg Syndrome* / genetics

Substances

  • SOX10 protein, human
  • SOXE Transcription Factors