Long QT interval in Turner syndrome--a high prevalence of LQTS gene mutations

PLoS One. 2013 Jul 25;8(7):e69614. doi: 10.1371/journal.pone.0069614. Print 2013.

Abstract

Objectives: QT-interval prolongation of unknown aetiology is common in Turner syndrome. This study set out to explore the presence of known long QT mutations in Turner syndrome and to examine the corrected QT-interval (QTc) over time and relate the findings to the Turner syndrome phenotype.

Methods: Adult women with Turner syndrome (n = 88) were examined thrice and 68 age-matched healthy controls were examined once. QTc was measured by one blinded reader (intra-reader variability: 0.7%), and adjusted for influence of heart rate by Bazett's (bQTc) and Hodges's formula (hQTc). The prevalence of mutations in genes related to Long QT syndrome was determined in women with Turner syndrome and a QTc >432.0 milliseconds (ms). Echocardiographic assessment of aortic valve morphology, 24-hour blood pressures and blood samples were done.

Results: The mean hQTc in women with Turner syndrome (414.0 ± 25.5 ms) compared to controls (390.4 ± 17.8 ms) was prolonged (p<0.001) and did not change over time (416.9 ± 22.6 vs. 415.6 ± 25.5 ms; p =0.4). 45,X karyotype was associated with increased hQTc prolongation compared to other Turner syndrome karyotypes (418.2 ± 24.8 vs. 407.6 ± 25.5 ms; p = 0.055). In women with Turner syndrome and a bQTc >432 ms, 7 had mutations in major Long QT syndrome genes (SCN5A and KCNH2) and one in a minor Long QT syndrome gene (KCNE2).

Conclusion: There is a high prevalence of mutations in the major LQTS genes in women with TS and prolonged QTc. It remains to be settled, whether these findings are related to the unexplained excess mortality in Turner women.

Clinical trial registration: NCT00624949. https://register.clinicaltrials.gov/prs/app/action/SelectProtocol/sid/S0001FLI/selectaction/View/ts/3/uid/U000099E.

Publication types

  • Clinical Trial
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aged
  • Aortic Valve / diagnostic imaging
  • Aortic Valve / pathology
  • Case-Control Studies
  • Death, Sudden, Cardiac / etiology
  • ERG1 Potassium Channel
  • Electrocardiography
  • Ether-A-Go-Go Potassium Channels / genetics*
  • Female
  • Genotype
  • Heart Rate
  • Humans
  • Karyotyping
  • Long QT Syndrome / complications
  • Long QT Syndrome / genetics*
  • Long QT Syndrome / mortality
  • Long QT Syndrome / physiopathology
  • Middle Aged
  • Mutation Rate
  • NAV1.5 Voltage-Gated Sodium Channel / genetics*
  • Potassium Channels, Voltage-Gated / genetics*
  • Survival Analysis
  • Turner Syndrome / complications
  • Turner Syndrome / genetics*
  • Turner Syndrome / mortality
  • Turner Syndrome / physiopathology
  • Ultrasonography

Substances

  • ERG1 Potassium Channel
  • Ether-A-Go-Go Potassium Channels
  • KCNE2 protein, human
  • KCNH2 protein, human
  • NAV1.5 Voltage-Gated Sodium Channel
  • Potassium Channels, Voltage-Gated
  • SCN5A protein, human

Associated data

  • ClinicalTrials.gov/NCT00624949

Grants and funding

Funding from the Danish Ministry for Science and Technology and Innovation, The Danish Heart Foundation, Novo Nordisk Foundation, Aase og Ejnar Danielsen Foundation, Korning Foundation, Hede Nielsens Foundation, Eva og Henry Frænkels Minde Foundation and Snedkermester Sophus Jacobsen og hustru Astrid Jacobsens Foundation. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.