Study of the PVRL1 gene in Italian nonsyndromic cleft lip patients with or without cleft palate

Ann Hum Genet. 2006 May;70(Pt 3):410-3. doi: 10.1111/j.1529-8817.2005.00237.x.

Abstract

Nonsyndromic cleft lip with or without cleft palate (CL/P) is a complex genetic trait and little is known about its aetiology. Recent investigations on rare clefting syndromes provided interesting clues about genes involved in face development. The PVRL1 gene encodes nectin1, a cell-to-cell adhesion molecule. Mutations in its sequence have been shown to cause the rare autosomal recessive syndrome CL/P-ectodermal dysplasia syndrome (CLPED1), while heterozygosity for the mutation W185X seemed to increase the risk of non syndromic CL/P in a population from northern Venezuela. In the present study, we screened 143 Italian CL/P patients for mutations in PVRL1. Three rare sequence variants in exon 3 that create amino-acid changes were detected in a total of 7 patients. Two of these mutations were not found in a panel of 292 unaffected controls, while the third was found in two controls. This study describes new mutations that may represent genetic risk factors for CL/P. Even though a study to look at the effects of the mutations on nectin1 function was not feasible, supporting evidence was reported, thus confirming the involvement of PVRL1 in the aetiology of non-syndromic CL/P malformation.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cell Adhesion Molecules / genetics*
  • Cleft Lip / complications
  • Cleft Lip / ethnology
  • Cleft Lip / genetics*
  • Cleft Palate / complications
  • Cleft Palate / ethnology
  • Cleft Palate / genetics*
  • DNA Mutational Analysis
  • Genetic Testing
  • Humans
  • Italy / ethnology
  • Mutation*
  • Nectins

Substances

  • Cell Adhesion Molecules
  • NECTIN1 protein, human
  • Nectins

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