Targeted gene panel sequencing for the rapid diagnosis of acutely ill infants

Mol Genet Genomic Med. 2019 Jul;7(7):e00796. doi: 10.1002/mgg3.796. Epub 2019 Jun 13.

Abstract

Background: Exome/genome sequencing (ES/GS) have been recently used in neonatal and pediatric/cardiac intensive care units (NICU and PICU/CICU) to diagnose and care for acutely ill infants, but the effectiveness of targeted gene panels for these purposes remains unknown.

Methods: RapSeq, a newly developed panel targeting 4,503 disease-causing genes, was employed on selected patients in our NICU/PICU/CICU. Twenty trios were sequenced from October 2015 to March 2017. We assessed diagnostic yield, turnaround times, and clinical consequences.

Results: A diagnosis was made in 10/20 neonates (50%); eight had de novo variants (ASXL1, CHD, FBN1, KMT2D, FANCB, FLNA, PAX3), one was a compound heterozygote for CHAT, and one had a maternally inherited GNAS variant. Preliminary reports were generated by 9.6 days (mean); final reports after Sanger sequencing at 16.3 days (mean). In all positive infants, the diagnosis changed management. In a case with congenital myasthenia, diagnosis and treatment occurred at 17 days versus 7 months in a historical control.

Conclusions: This study shows that a gene panel that includes the majority of known disease-causing genes can rapidly identify a diagnosis in a large number of tested infants. Due to simpler deployment and interpretation and lower costs, this approach might represent an alternative to ES/GS in the NICU/PICU/CICU.

Keywords: genetic diagnosis; neonatology; newborn; precision medicine; rapid sequencing.

MeSH terms

  • Diagnosis
  • Diagnostic Techniques and Procedures
  • Disease / genetics*
  • Early Diagnosis*
  • Exome
  • Exome Sequencing
  • Female
  • Genetic Testing / economics
  • Genetic Testing / methods*
  • Genetic Testing / trends
  • Humans
  • Infant
  • Infant, Newborn
  • Intensive Care Units, Neonatal / trends
  • Male