Oligodontia is caused by mutation in LTBP3, the gene encoding latent TGF-beta binding protein 3

Am J Hum Genet. 2009 Apr;84(4):519-23. doi: 10.1016/j.ajhg.2009.03.007. Epub 2009 Apr 2.

Abstract

We have identified a consanguineous Pakistani family where oligodontia is inherited along with short stature in an autosomal-recessive fashion. Increased bone density was present in the spine and at the base of the skull. Using high-density single-nucleotide polymorphism microarrays for homozygosity mapping, we identified a 28 Mb homozygous stretch shared between affected individuals on chromosome 11q13. Screening selected candidate genes within this region, we identified a homozygous nonsense mutation, Y774X, within LTBP3, the gene for the latent TGF-beta binding protein 3, an extracellular matrix protein believed to be required for osteoclast function.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Anodontia / genetics*
  • Anodontia / metabolism
  • Anodontia / pathology
  • Base Sequence
  • Body Height / genetics
  • Bone Density / genetics
  • Bone Development / genetics
  • Chromosomes, Human, Pair 11 / genetics
  • Codon, Nonsense*
  • Consanguinity
  • DNA Primers / genetics
  • Female
  • Heterozygote
  • Homozygote
  • Humans
  • Latent TGF-beta Binding Proteins / genetics*
  • Male
  • Oligonucleotide Array Sequence Analysis
  • Pakistan
  • Pedigree
  • Phenotype
  • RNA, Messenger / genetics
  • RNA, Messenger / metabolism

Substances

  • Codon, Nonsense
  • DNA Primers
  • LTBP3 protein, human
  • Latent TGF-beta Binding Proteins
  • RNA, Messenger