Putting genome-wide sequencing in neonates into perspective

Genet Med. 2019 May;21(5):1074-1082. doi: 10.1038/s41436-018-0293-0. Epub 2018 Oct 5.

Abstract

Purpose: Several studies have reported diagnostic yields up to 57% for rapid exome or genome sequencing (rES/GS) as a single test in neonatal intensive care unit (NICU) patients, but the additional yield of rES/GS compared with other available diagnostic options still remains unquantified in this population.

Methods: We retrospectively evaluated all genetic NICU consultations in a 2-year period.

Results: In 132 retrospectively evaluated NICU consultations 27 of 32 diagnoses (84.4%) were made using standard genetic workup. Most diagnoses (65.6%) were made within 16 days. Diagnostic ES yield was 5/29 (17.2%). Genetic diagnoses had a direct effect on clinical management in 90.6% (29/32) of patients.

Conclusions: Our study shows that exome sequencing has a place in NICU diagnostics, but given the associated costs and the high yield of alternative diagnostic strategies, we recommend to first perform clinical genetic consultation.

Keywords: ES; NICU; clinical geneticists; rapid; sequencing.

MeSH terms

  • Chromosome Mapping / methods
  • Exome / genetics
  • Exome Sequencing / economics
  • Exome Sequencing / methods
  • Female
  • Genetic Testing / economics
  • Genome-Wide Association Study / methods
  • Humans
  • Infant, Newborn
  • Infant, Newborn, Diseases / diagnosis*
  • Infant, Newborn, Diseases / genetics*
  • Intensive Care, Neonatal
  • Male
  • Retrospective Studies