Investigative Ophthalmology & Visual Science Cover Image for Volume 65, Issue 7
June 2024
Volume 65, Issue 7
Open Access
ARVO Annual Meeting Abstract  |   June 2024
An intronic tandem repeat expansion in TMCO1 is associated with open angle glaucoma severity
Author Affiliations & Notes
  • Owen Siggs
    Garvan Institute of Medical Research, Darlinghurst, New South Wales, Australia
  • Shannon Ji
    Garvan Institute of Medical Research, Darlinghurst, New South Wales, Australia
  • Joshua Schmidt
    Flinders University, Adelaide, South Australia, Australia
  • Yves Fontaine
    Garvan Institute of Medical Research, Darlinghurst, New South Wales, Australia
  • David Dimasi
    Flinders University, Adelaide, South Australia, Australia
  • Jamie E Craig
    Flinders University, Adelaide, South Australia, Australia
  • Footnotes
    Commercial Relationships   Owen Siggs Seonix Bio, Code I (Personal Financial Interest), Seonix Bio, Code O (Owner), Seonix Bio, Code S (non-remunerative); Shannon Ji None; Joshua Schmidt None; Yves Fontaine None; David Dimasi Seonix Bio, Code E (Employment); Jamie Craig Seonix Bio, Code I (Personal Financial Interest), Seonix Bio, Code O (Owner), Seonix Bio, Code S (non-remunerative)
  • Footnotes
    Support  National Health and Medical Research Council; Snow Medical
Investigative Ophthalmology & Visual Science June 2024, Vol.65, 2168. doi:
  • Views
  • Share
  • Tools
    • Alerts
      ×
      This feature is available to authenticated users only.
      Sign In or Create an Account ×
    • Get Citation

      Owen Siggs, Shannon Ji, Joshua Schmidt, Yves Fontaine, David Dimasi, Jamie E Craig; An intronic tandem repeat expansion in TMCO1 is associated with open angle glaucoma severity. Invest. Ophthalmol. Vis. Sci. 2024;65(7):2168.

      Download citation file:


      © ARVO (1962-2015); The Authors (2016-present)

      ×
  • Supplements
Abstract

Purpose : Glaucoma is one of the most heritable human diseases, with variants at the TMCO1 locus among the strongest common genetic risk factors known. This risk has recently been attributed to expansion of an intronic 28bp variable number tandem repeat (VNTR) in TMCO1. We sought to understand the clinical impact of TMCO1 VNTR expansion length in a glaucoma registry.

Methods : 3123 participants were genotyped at two variants associated with TMCO1 VNTR expansions of increasing lengths. In a subset of individuals, repeat lengths were also measured by a targeted PCR-based assay, by short-read whole genome sequencing, or by nanopore long-read sequencing. VNTR lengths were then associated with a range of clinical outcomes.

Results : Increased TMCO1 VNTR length was associated in a dose-dependent manner with a younger age at glaucoma diagnosis, higher prevalence of high-tension glaucoma, higher IOP, and a higher likelihood of glaucoma family history. The clinical phenotype of individuals with the longest TMCO1 repeat expansions was similar to that of MYOC p.Gln368Ter heterozygotes, although at a 3.5 times higher frequency in the general population.

Conclusions : TMCO1 VNTR expansions represent a new class of genetic risk variant in glaucoma, with longer expansions associated with earlier-onset and more severe disease. The molecular mechanism by which these expansions increase glaucoma risk remains to be determined.

This abstract was presented at the 2024 ARVO Annual Meeting, held in Seattle, WA, May 5-9, 2024.

×
×

This PDF is available to Subscribers Only

Sign in or purchase a subscription to access this content. ×

You must be signed into an individual account to use this feature.

×