A fast and cost-effective molecular diagnostic tool for genetic diseases involved in sudden cardiac death

Clin Chim Acta. 2016 Jan 30:453:80-5. doi: 10.1016/j.cca.2015.12.011. Epub 2015 Dec 10.

Abstract

Background: Cardiomyopathies and arrhythmia syndromes are common genetic cardiac diseases that account for a significant number of sudden cardiac death (SCD) cases.

Methods: NGS workflow based on a panel of 95 genes was developed on Illumina NextSeq500™ sequencer for sequencing prevalent SCD-causing genes. A cohort of 90 patients (56 genotype-positive, 27 genotype-negative and 7 new cases) was screened to evaluate this strategy in terms of sensitivity, specificity, practicability and cost. In silico analysis were performed using a pipeline based on NextGENe® software and a personalized Sophia Genetics pipeline.

Results: Using our panel custom, 100% of targeted sequences were efficiently covered and all previously identified genetic variants were readily detected. Applied to 27 genotype-negative patients, this molecular strategy allowed the identification of pathogenic or likely pathogenic variants into 12 cases. It confirmed the involvement of HCN4 mutations in the combined bradycardia–myocardial non-compaction phenotype, and also suggested, for the first time, the involvement of PKP2, usually associated with arrhythmogenic right ventricular dysplasia, in ventricular non-compaction.

Conclusion: This NGS approach is a fast, cheap, sensitive and high-throughput mutation detection method that is ready to be deployed in clinical laboratories and would provide new insights on physiopathology of SCD, more particularly of cardiomyopathies and arrhythmia syndromes.

Keywords: Arrhythmia syndromes; Cardiomyopathies; Molecular diagnosis; Mutations; NGS sequencing; Sudden cardiac death.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cost-Benefit Analysis*
  • Death, Sudden, Cardiac*
  • Exons / genetics
  • Genotype
  • Humans
  • Introns / genetics
  • Molecular Diagnostic Techniques / economics*
  • Time Factors