Epigenomic newborn screening for conditions with intellectual disability and autistic features in Australian newborns

Epigenomics. 2024;16(18):1203-1214. doi: 10.1080/17501911.2024.2402681. Epub 2024 Oct 4.

Abstract

This study describes a protocol to assess a novel workflow called Epi-Genomic Newborn Screening (EpiGNs) on 100,000 infants from the state of Victoria, Australia. The workflow uses a first-tier screening approach called methylation-specific quantitative melt analysis (MS-QMA), followed by second and third tier testing including targeted methylation and copy number variation analyzes with droplet digital PCR, EpiTYPER system and low-coverage whole genome sequencing. EpiGNs utilizes only two 3.2 mm newborn blood spot punches to screen for genetic conditions, including fragile X syndrome, Prader-Willi syndrome, Angelman syndrome, Dup15q syndrome and sex chromosome aneuploidies. The program aims to: identify clinically actionable methylation screening thresholds for the first-tier screen and estimate prevalence for the conditions screened.

Keywords: EpiGNs; early detection; epigenomics; newborn screening; population-wide study.

Plain language summary

[Box: see text].

MeSH terms

  • Aneuploidy
  • Angelman Syndrome / diagnosis
  • Angelman Syndrome / genetics
  • Australia
  • Autistic Disorder / diagnosis
  • Autistic Disorder / genetics
  • Chromosome Duplication
  • Chromosomes, Human, Pair 15 / genetics
  • DNA Copy Number Variations
  • DNA Methylation*
  • Epigenomics / methods
  • Female
  • Fragile X Syndrome / diagnosis
  • Fragile X Syndrome / genetics
  • Genetic Testing / methods
  • Humans
  • Infant, Newborn
  • Intellectual Disability / diagnosis
  • Intellectual Disability / genetics
  • Male
  • Neonatal Screening* / methods
  • Prader-Willi Syndrome* / diagnosis
  • Prader-Willi Syndrome* / genetics