Identification of known and unknown genes associated with mitral valve prolapse using an exome slice methodology

J Med Genet. 2020 Dec;57(12):843-850. doi: 10.1136/jmedgenet-2019-106715. Epub 2020 Apr 10.

Abstract

Purpose: Although a familial distribution has been documented, the genetic aetiology of mitral valve prolapse (MVP) is largely unknown, with only four genes identified so far: FLNA, DCHS1, DZIP1 and PLD1. The aim of this study was to evaluate the genetic yield in known causative genes and to identify possible novel genes associated with MVP using a heart gene panel based on exome sequencing.

Methods: Patients with MVP were referred for genetic counselling when a positive family history for MVP was reported and/or Barlow's disease was diagnosed. In total, 101 probands were included to identify potentially pathogenic variants in a set of 522 genes associated with cardiac development and/or diseases.

Results: 97 (96%) probands were classified as Barlow's disease and 4 (4%) as fibroelastic deficiency. Only one patient (1%) had a likely pathogenic variant in the known causative genes (DCHS1). However, an interesting finding was that 10 probands (11%) had a variant that was classified as likely pathogenic in six different, mostly cardiomyopathy genes: DSP (1×), HCN4 (1×), MYH6 (1×), TMEM67 (1×), TRPS1 (1×) and TTN (5×).

Conclusion: Exome slice sequencing analysis performed in MVP probands reveals a low genetic yield in known causative genes but may expand the cardiac phenotype of other genes. This study suggests for the first time that also genes related to cardiomyopathy may be associated with MVP. This highlights the importance to screen these patients and their family for the presence of arrhythmias and of 'disproportionate' LV remodelling as compared with the severity of mitral regurgitation, unravelling a possible coexistent cardiomyopathy.

Keywords: clinical genetics; valvar diseases.

MeSH terms

  • Adaptor Proteins, Signal Transducing / genetics
  • Aged
  • Cardiac Myosins / genetics
  • Cardiomyopathies / genetics*
  • Cardiomyopathies / pathology
  • Connectin
  • Desmoplakins / genetics
  • Exome / genetics
  • Exome Sequencing
  • Female
  • Genetic Association Studies*
  • Genetic Predisposition to Disease*
  • Humans
  • Hyperpolarization-Activated Cyclic Nucleotide-Gated Channels / genetics
  • Male
  • Membrane Proteins / genetics
  • Middle Aged
  • Mitral Valve Prolapse / genetics*
  • Mitral Valve Prolapse / pathology
  • Muscle Proteins / genetics
  • Myosin Heavy Chains / genetics
  • Pedigree
  • Potassium Channels / genetics
  • Repressor Proteins / genetics

Substances

  • Adaptor Proteins, Signal Transducing
  • Connectin
  • DSP protein, human
  • DZIP1 protein, human
  • Desmoplakins
  • HCN4 protein, human
  • Hyperpolarization-Activated Cyclic Nucleotide-Gated Channels
  • MYH6 protein, human
  • Membrane Proteins
  • Muscle Proteins
  • Potassium Channels
  • Repressor Proteins
  • TMEM67 protein, human
  • TRPS1 protein, human
  • TTN protein, human
  • Cardiac Myosins
  • Myosin Heavy Chains

Supplementary concepts

  • Barlow syndrome