Clinical and molecular characterization of a patient with an interstitial deletion of chromosome 12q15-q23 and peripheral corneal abnormalities

Am J Ophthalmol. 2006 Mar;141(3):566-567. doi: 10.1016/j.ajo.2005.09.024.

Abstract

Purpose: To describe the ocular features of a patient with an interstitial deletion of chromosome 12 and to determine the molecular boundaries of the deletion.

Design: Observational case report and laboratory investigation.

Methods: A patient with an interstitial deletion of chromosome 12 was clinically examined for ocular abnormalities. DNA samples were used for molecular studies to define the deletion boundaries.

Results: Ocular examination showed abnormalities of the anterior segment consistent with a diagnosis of cornea plana. Molecular analyses showed the deletion included the KERA gene, the SLRP (small leucine repeat protein) gene cluster, the genetic loci for autosomal-dominant (CNA1) and autosomal-recessive (CNA2) cornea plana, and a portion of the mapped locus for high myopia (MYP3).

Conclusions: These results, combined with previous genetic linkage studies, identifies a 3-cM region located between microsatellite markers D12S82 and D12S351 that is likely to contain a gene responsible for CNA1.

Publication types

  • Case Reports
  • Research Support, N.I.H., Extramural

MeSH terms

  • Adult
  • Calcineurin / genetics
  • Carrier Proteins / genetics
  • Child, Preschool
  • Chromosome Deletion*
  • Chromosomes, Human, Pair 12 / genetics*
  • Cornea / abnormalities*
  • Cornea / pathology
  • Corneal Dystrophies, Hereditary / genetics*
  • Corneal Dystrophies, Hereditary / pathology
  • Female
  • Genetic Linkage
  • Humans
  • Male
  • Membrane Proteins / genetics
  • Microsatellite Repeats
  • Myopia / genetics
  • Proteoglycans / genetics

Substances

  • Carrier Proteins
  • KERA protein, human
  • LRRC1 protein, human
  • Membrane Proteins
  • Proteoglycans
  • Calcineurin