The human T locus and spina bifida risk

Hum Genet. 2004 Nov;115(6):475-82. doi: 10.1007/s00439-004-1185-8. Epub 2004 Sep 24.

Abstract

The transcription factor T is essential for mesoderm formation and axial development during embryogenesis. Embryonic genotype for a single-nucleotide polymorphism in intron 7 of T ( TIVS7 T/C) has been associated with the risk of spina bifida in some but not all studies. We developed a novel genotyping assay for the TIVS7 polymorphism using heteroduplex generator methodology. This assay was used to genotype spina bifida case-parent trios and the resulting data were analyzed using the transmission disequilibrium test and log-linear analyses. Analyses of these data demonstrated that heterozygous parents transmit the TIVS7-C allele to their offspring with spina bifida significantly more frequently than expected under the assumption of Mendelian inheritance (63 vs 50%, P=0.02). Moreover, these analyses suggest that the TIVS7-C allele acts in a dominant fashion, such that individuals carrying one or more copies of this allele have a 1.6-fold increased risk of spina bifida compared with individuals with zero copies. In silico analysis of the sequence surrounding this polymorphism revealed a potential target site for olfactory neuron-specific factor-1, a transcription factor expressed in the neural tube during development, spanning the polymorphic site. Several other putative, developmentally important and/or environmentally responsive transcription factor-binding sites were also identified close to the TIVS7 polymorphism. The TIVS7 polymorphism or a variant that is in linkage disequilibrium with the TIVS7 polymorphism may, therefore, play a role in T gene expression and influence the risk of spina bifida.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Alleles
  • Base Sequence
  • Binding Sites
  • Databases as Topic
  • Genes, Dominant
  • Genetic Techniques*
  • Genotype
  • Heterozygote
  • Humans
  • Introns
  • Linkage Disequilibrium
  • Models, Genetic
  • Models, Statistical
  • Molecular Sequence Data
  • Mutagenesis, Site-Directed
  • Nucleic Acid Heteroduplexes
  • Polymorphism, Genetic
  • Polymorphism, Single Nucleotide
  • Risk
  • Spinal Dysraphism / genetics*

Substances

  • Nucleic Acid Heteroduplexes