GWAS reveals new recessive loci associated with non-syndromic facial clefting

Eur J Med Genet. 2012 Oct;55(10):510-4. doi: 10.1016/j.ejmg.2012.06.005. Epub 2012 Jun 27.

Abstract

We have applied a GWAS to 40 consanguineous families segregating cases of non-syndromic cleft lip with or without cleft palate (NS CL/P) (a total of 160 affected and unaffected individuals) in order to trace potential recessive loci that confer susceptibility to this common facial malformation. Pedigree-based association test (PBAT) analyses reported nominal evidence of association and linkage over SNP markers located at 11q25 (rs4937877, P = 2.7 × 10(-6)), 19p12 (rs4324267, P = 1.6 × 10(-5)), 5q14.1 (rs4588572, P-value = 3.36 × 10(-5)), and 15q21.1 (rs4774497, P = 1.08 × 10(-4)). Using the Versatile Gene-Based Association Study to complement the PBAT results, we found clusters of markers located at chromosomes 19p12, 11q25, and 8p23.2 overcome the threshold for GWAS significance (P < 1 × 10(-7)). From this study, new recessive loci implicated in NS CL/P include: B3GAT1, GLB1L2, ZNF431, ZNF714, and CSMD1, even though the functional association with the genesis of NS CL/P remains to be elucidated. These results emphasize the importance of using homogeneous populations, phenotypes, and family structures for GWAS combined with gene-based association analyses, and should encourage. other researchers to evaluate these genes on independent patient samples affected by NS CL/P.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Brain / abnormalities
  • Case-Control Studies
  • Cleft Lip / epidemiology
  • Cleft Lip / genetics*
  • Cleft Palate / epidemiology
  • Cleft Palate / genetics*
  • DNA-Binding Proteins / genetics
  • Female
  • Genes, Recessive*
  • Genetic Markers
  • Genetic Predisposition to Disease / genetics
  • Genome-Wide Association Study
  • Glucuronosyltransferase / genetics
  • Glycoside Hydrolases / genetics
  • Humans
  • Male
  • Membrane Proteins / genetics
  • Pedigree
  • Transcription Factors / genetics
  • Tumor Suppressor Proteins

Substances

  • CSMD1 protein, human
  • DNA-Binding Proteins
  • Genetic Markers
  • Membrane Proteins
  • Transcription Factors
  • Tumor Suppressor Proteins
  • ZNF431 protein, human
  • ZNF714 protein, human
  • galactosylgalactoylxylosylprotein 3-beta-glucuronosyltransferase
  • Glucuronosyltransferase
  • Glycoside Hydrolases
  • GLB1L2 protein, human

Supplementary concepts

  • Orofacial Cleft 1