Vortex Pattern of Corneal Deposits in Granular Corneal Dystrophy Associated With the p.(Arg555Trp) Mutation in TGFBI

Cornea. 2017 Feb;36(2):210-216. doi: 10.1097/ICO.0000000000001045.

Abstract

Purpose: To describe 2 unrelated families with multiple members demonstrating a less commonly recognized vortex pattern of corneal deposits confirmed to be granular corneal dystrophy type 1 (GCD1) after identification of the p.(Arg555Trp) mutation in the transforming growth factor β-induced gene (TGFBI).

Methods: A slit-lamp examination was performed on individuals from 2 families, one of Mexican descent and a second of Italian descent. After DNA extraction from affected individuals and their unaffected relatives, TGFBI screening was performed.

Results: Eight of 20 individuals in the Mexican family and 20 of 55 in the Italian family demonstrated corneal stromal opacities. Seven of the 8 affected individuals in the Mexican family and 4 of the 20 affected individuals in the Italian family demonstrated a phenotype characterized by a "sea fan" or vortex pattern of superficial stromal corneal deposits originating from the inferior aspect of the cornea. Screening of TGFBI in both families revealed a heterozygous missense mutation [p.(Arg555Trp)] in exon 12, confirming the diagnosis of GCD1.

Conclusions: Our findings demonstrate that GCD1 may present with a vortex pattern of anterior stromal deposits. Although this pattern of dystrophic deposits is not recognized by clinicians as a typical phenotype of GCD1, it is consistent with the production of the majority of the TGFBI protein by the corneal epithelium.

MeSH terms

  • Adolescent
  • Aged, 80 and over
  • Child
  • Corneal Dystrophies, Hereditary / diagnosis*
  • Corneal Dystrophies, Hereditary / genetics*
  • Corneal Opacity / diagnosis
  • Corneal Stroma / pathology*
  • DNA Mutational Analysis
  • Extracellular Matrix Proteins / genetics*
  • Female
  • Gene Frequency
  • Heterozygote
  • Humans
  • Italy
  • Male
  • Mexico
  • Middle Aged
  • Mutation, Missense*
  • Pedigree
  • Polymerase Chain Reaction
  • Slit Lamp
  • Transforming Growth Factor beta / genetics*
  • Young Adult

Substances

  • Extracellular Matrix Proteins
  • Transforming Growth Factor beta
  • betaIG-H3 protein

Supplementary concepts

  • Groenouw type I corneal dystrophy