Analysis of FKBP10, SERPINH1, and SERPINF1 genes in patients with osteogenesis imperfecta

Genet Mol Res. 2016 Sep 2;15(3). doi: 10.4238/gmr.15038665.

Abstract

Osteogenesis imperfecta (OI) is a heterogeneous disorder that causes fragility, deformity, and fractures in bones. A large number of genes that are associated with the disease have been identified in the last decade; this makes the genetic diagnosis of OI more difficult. To improve our knowledge of the genetic mutation profile in OI we used single-stranded conformation polymorphism screening and automated sequencing to investigate the SERPINH1, FKBP10, and SERPINF1 genes, which are related to recessive OI, in 23 unrelated Brazilian patients. Nine rare changes and four common polymorphisms were detected. Most changes were benign genetic variants. In general, changes in the SERPINH1 and SERPINF1 genes were synonymous polymorphisms or missense changes located in non-coding regions. A pathogenic change was found in the FKBP10 gene. The characterization of mutations related to OI in distinct populations can improve our knowledge of the genetic aspects of OI and help us develop molecular strategies for the diagnosis of the disease.

MeSH terms

  • Base Sequence
  • Bone and Bones / metabolism
  • Bone and Bones / pathology
  • Brazil
  • Case-Control Studies
  • Eye Proteins / genetics*
  • Eye Proteins / metabolism
  • Gene Expression
  • Genes, Recessive
  • HSP47 Heat-Shock Proteins / genetics*
  • HSP47 Heat-Shock Proteins / metabolism
  • Humans
  • Mutation
  • Nerve Growth Factors / genetics*
  • Nerve Growth Factors / metabolism
  • Osteogenesis Imperfecta / genetics*
  • Osteogenesis Imperfecta / metabolism
  • Osteogenesis Imperfecta / pathology
  • Polymorphism, Single-Stranded Conformational*
  • Serpins / genetics*
  • Serpins / metabolism
  • Tacrolimus Binding Proteins / genetics*
  • Tacrolimus Binding Proteins / metabolism

Substances

  • Eye Proteins
  • HSP47 Heat-Shock Proteins
  • Nerve Growth Factors
  • SERPINH1 protein, human
  • Serpins
  • pigment epithelium-derived factor
  • Tacrolimus Binding Proteins
  • FKBP10 protein, human