Frequency of CEP290 c.2991_1655A>G mutation in 175 Spanish families affected with Leber congenital amaurosis and early-onset retinitis pigmentosa

Mol Vis. 2007 Nov 27:13:2160-2.

Abstract

Purpose: Leber congenital amaurosis (LCA) is the most severe inherited retinopathy with the earliest age of onset. To date, eleven genes have been reported to cause the non-syndromic LCA phenotype. The CEP290 gene has been shown to account for Joubert and Senior-Loken syndromes and to represent a frequent cause of non-syndromic LCA. The aim of the present study was to establish the prevalence of CEP290 c.2991_1655A>G in non-syndromic Spanish patients having LCA or early-onset retinitis pigmentosa (RP).

Methods: We used automated sequencing to examine 49 non-syndromic Spanish families with LCA and 126 Spanish families with early-onset RP for the CEP290 c.2991_1655A>G mutation. As a control, we recruited 50 unrelated Spanish healthy individuals.

Results: The frequencies of mutated alleles were 6% in LCA cases and 0% in early-onset RP and healthy individual controls. These results were compared to other populations.

Conclusions: The CEP290 c.2991_1655A>G mutation frequency in Spanish non-syndromic LCA families is lower than that of other countries.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adenine
  • Age of Onset
  • Antigens, Neoplasm / genetics*
  • Blindness / congenital
  • Blindness / etiology
  • Blindness / genetics*
  • Cell Cycle Proteins
  • Cohort Studies
  • Cytoskeletal Proteins
  • Gene Frequency*
  • Guanine
  • Humans
  • Mutation*
  • Neoplasm Proteins / genetics*
  • Phenotype
  • Retinal Diseases / complications
  • Retinal Diseases / genetics*
  • Retinitis Pigmentosa / epidemiology
  • Retinitis Pigmentosa / genetics*
  • Spain

Substances

  • Antigens, Neoplasm
  • Cell Cycle Proteins
  • Cep290 protein, human
  • Cytoskeletal Proteins
  • Neoplasm Proteins
  • Guanine
  • Adenine