Cockayne syndrome caused by paternally inherited 5 Mb deletion of 10q11.2 and a frameshift mutation of ERCC6

Eur J Med Genet. 2011 May-Jun;54(3):272-6. doi: 10.1016/j.ejmg.2011.02.008. Epub 2011 Mar 2.

Abstract

Cockayne syndrome is a rare multi-systemic autosomal recessive condition characterized by variable post natal growth failure, neurological impairment, feeding difficulty, and progressive skin, ophthalmological, auditory and dental abnormalities. Life-span is usually shortened and death occurs in childhood or adolescence in the majority of cases. Only 3 cases of chromosomal aberrations as a cause of CS have been previously reported. We report a patient with a clinical phenotype of severe infantile CS who has a paternally inherited 5 Mb deletion of 10q11.2 resulting in loss of one allele and a previously unreported frameshift mutation of ERCC6 on the maternal allele.

Publication types

  • Case Reports

MeSH terms

  • Child, Preschool
  • Chromosome Banding
  • Chromosome Deletion*
  • Chromosomes, Human, Pair 10 / genetics*
  • Cockayne Syndrome / genetics*
  • Cockayne Syndrome / pathology
  • DNA Helicases / genetics*
  • DNA Mutational Analysis
  • DNA Repair Enzymes / genetics*
  • Fatal Outcome
  • Female
  • Frameshift Mutation*
  • Humans
  • Infant
  • Poly-ADP-Ribose Binding Proteins

Substances

  • Poly-ADP-Ribose Binding Proteins
  • DNA Helicases
  • ERCC6 protein, human
  • DNA Repair Enzymes