A novel mutation in transforming growth factor-beta induced protein (TGFβIp) reveals secondary structure perturbation in lattice corneal dystrophy

Br J Ophthalmol. 2011 Oct;95(10):1457-62. doi: 10.1136/bjophthalmol-2011-300651. Epub 2011 Aug 10.

Abstract

Background: To describe mutations in the transforming growth factor-beta induced (TGFBI) gene in Asian patients with Bowman's membrane as well as stromal corneal dystrophies, and to elucidate their structural implications, using model peptides.

Methods: Twenty-two unrelated Asian families were examined clinically including visual acuity testing and ocular examination with slit lamp biomicroscopy. Genomic DNA was extracted and the 17 exons of the TGFBI gene were amplified by PCR and sequenced bi-directionally. Biophysical techniques were used to characterise the wild type and mutant model peptides.

Results: Molecular genetic analysis identified a variety of mutations in our patient series including a novel heterozygous C to A transversion mutation in exon 14 (c.1859C→A), resulting in a substitution of a highly conserved alanine residue by aspartic acid (p.A620D). Clinical presentation in the patient with the p.A620D included subepithelial scarring in addition to the linear branching opacities usually seen with lattice dystrophy. Using model peptides, we showed that A620D mutant peptide alters the secondary structure and conformational stability, and increased amyloid formation.

Conclusion: A novel mutation (A620D) in transforming growth factor-beta induced protein (TGFβIp) is described, expanding the repertoire of mutations in this protein. Using model peptides, we demonstrated that A→D substitution leads to perturbation of the secondary structure that may be responsible for the amyloid formation in lattice corneal dystrophy.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Amino Acid Sequence
  • Amyloid / metabolism
  • Amyloid / ultrastructure
  • Asian People / genetics
  • Benzothiazoles
  • Bowman Membrane / pathology
  • Child
  • Circular Dichroism
  • Contrast Sensitivity / physiology
  • Corneal Dystrophies, Hereditary / genetics*
  • Corneal Dystrophies, Hereditary / pathology
  • Corneal Stroma / pathology
  • Crystallography, X-Ray
  • Extracellular Matrix Proteins / genetics*
  • Female
  • Humans
  • Male
  • Middle Aged
  • Molecular Sequence Data
  • Mutation*
  • Polymerase Chain Reaction
  • Refraction, Ocular / physiology
  • Thiazoles
  • Transforming Growth Factor beta / genetics*
  • Visual Acuity / physiology

Substances

  • Amyloid
  • Benzothiazoles
  • Extracellular Matrix Proteins
  • Thiazoles
  • Transforming Growth Factor beta
  • betaIG-H3 protein
  • thioflavin T