Coincidental Occurrence of Schnyder Corneal Dystrophy and Posterior Polymorphous Corneal Dystrophy Type 3

Cornea. 2019 Jun;38(6):758-760. doi: 10.1097/ICO.0000000000001930.

Abstract

Purpose: To report a simultaneous occurrence of 2 rare corneal dystrophies.

Methods: A 30-year-old man with a family history of posterior polymorphous corneal dystrophy type 3 (PPCD3) was invited for ophthalmic examination. Sanger sequencing of the coding regions and intron/exon boundaries of disease-associated genes, ZEB1 and UBIAD1, was performed.

Results: The clinical findings suggested co-occurrence of PPCD3 and Schnyder corneal dystrophy in the proband. This dual diagnosis was supported by genetic findings. He was identified to carry a previously reported heterozygous nonsense mutation in ZEB1: c.2157C>G; p.(Tyr719*), and a novel heterozygous missense mutation in UBIAD1: c.569T>C; p.(Ile190Thr). The mother of the proband only carried c.2157C>G in ZEB1, and slit-lamp examination of her corneas showed endothelial lesions characteristic of PPCD3. The sister of the proband carried c.569T>C in UBIAD1 and had corneal crystal deposition in her anterior stroma consistent with the diagnosis of Schnyder corneal dystrophy.

Conclusions: This case illustrates the coincidental occurrence of 2 rare and genetically distinct corneal dystrophies in a single patient. Furthermore, it highlights the need to perform comprehensive phenotyping in combination with appropriate genetic diagnostic testing to achieve an accurate diagnosis.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Corneal Dystrophies, Hereditary / diagnosis*
  • Corneal Dystrophies, Hereditary / genetics
  • Dimethylallyltranstransferase / genetics
  • Humans
  • Male
  • Mutation
  • Zinc Finger E-box-Binding Homeobox 1 / genetics

Substances

  • ZEB1 protein, human
  • Zinc Finger E-box-Binding Homeobox 1
  • Dimethylallyltranstransferase
  • UBIAD1 protein, human

Supplementary concepts

  • Corneal Dystrophy, Crystalline, of Schnyder
  • Corneal Dystrophy, Posterior Polymorphous, 3