Clinical utility of exome sequencing in infantile heart failure

Genet Med. 2020 Feb;22(2):423-426. doi: 10.1038/s41436-019-0654-3. Epub 2019 Sep 17.

Abstract

Purpose: Pediatric cardiomyopathy is rare, has a broad differential diagnosis, results in high morbidity and mortality, and has suboptimal diagnostic yield using next-generation sequencing panels. Exome sequencing has reported diagnostic yields ranging from 30% to 57% for neonates in intensive care units. We aimed to characterize the clinical utility of exome sequencing in infantile heart failure.

Methods: Infants diagnosed with acute heart failure prior to 1 year old over a period of 34 months at a large tertiary children's hospital were recruited. Demographic and diagnostic information was obtained from medical records. Fifteen eligible patients were enrolled.

Results: Dilated cardiomyopathy was the predominant cardiac diagnosis, seen in 60% of patients. A molecular diagnosis was identified in 66.7% of patients (10/15). Of those diagnoses, 70% would not have been detected using multigene next-generation sequencing panels focused on cardiomyopathy or arrhythmia disease genes. Genetic testing changed medical decision-making in 53% of all cases and 80% of positive cases, and was especially beneficial when testing was expedited.

Conclusion: Given the broad differential diagnosis and critical status of infants with heart failure, rapid exome sequencing provides timely diagnoses, changes medical management, and should be the first-tier molecular test.

Keywords: cardiomyopathy; clinical utility; exome sequencing; heart failure; pediatric.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Cardiomyopathies / genetics
  • Cardiomyopathy, Dilated / genetics
  • Exome / genetics
  • Exome Sequencing / methods
  • Exome Sequencing / trends*
  • Female
  • Genetic Testing / ethics*
  • Genetic Testing / trends
  • Heart Failure / diagnosis*
  • Heart Failure / genetics
  • High-Throughput Nucleotide Sequencing / methods
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Treatment Outcome