Identification of three novel mutations in the CHD7 gene in patients with clinical signs of typical or atypical CHARGE syndrome

Int J Pediatr Otorhinolaryngol. 2010 Dec;74(12):1441-4. doi: 10.1016/j.ijporl.2010.09.006. Epub 2010 Oct 12.

Abstract

CHARGE syndrome is an autosomal dominant disorder characterized by features represented in its acronym: Coloboma, Heart defect, Atresia of the choanae, Retarded growth and development, Genital abnormalities, Ear anomalies/deafness. We report two patients with a diagnosis of typical CHARGE syndrome and one with atypical clinical diagnosis. All the three patients had uni- or bilateral choanal atresia and sensorineural hearing loss. The patients were screened for CHD7 gene mutations. Three novel occurring de novo heterozygous mutations were identified: a mutation in the donor splice site of intron 24, a missense mutation in exon 2 and a deletion in exon 11.

Publication types

  • Case Reports

MeSH terms

  • CHARGE Syndrome / diagnosis
  • CHARGE Syndrome / genetics*
  • DNA Helicases / genetics*
  • DNA-Binding Proteins / genetics*
  • Diseases in Twins / genetics
  • Female
  • Heterozygote
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Mutation*
  • Sequence Analysis, DNA

Substances

  • DNA-Binding Proteins
  • DNA Helicases
  • CHD7 protein, human