Targeted next-generation sequencing to diagnose disorders of HDL cholesterol

J Lipid Res. 2015 Oct;56(10):1993-2001. doi: 10.1194/jlr.P058891. Epub 2015 Aug 8.

Abstract

A low level of HDL cholesterol (HDL-C) is a common clinical scenario and an important marker for increased cardiovascular risk. Many patients with very low or very high HDL-C have a rare mutation in one of several genes, but identification of the molecular abnormality in patients with extreme HDL-C is rarely performed in clinical practice. We investigated the accuracy and diagnostic yield of a targeted next-generation sequencing (NGS) assay for extreme levels of HDL-C. We developed a targeted NGS panel to capture the exons, intron/exon boundaries, and untranslated regions of 26 genes with highly penetrant effects on plasma lipid levels. We sequenced 141 patients with extreme HDL-C levels and prioritized variants in accordance with medical genetics guidelines. We identified 35 pathogenic and probably pathogenic variants in HDL genes, including 21 novel variants, and performed functional validation on a subset of these. Overall, a molecular diagnosis was established in 35.9% of patients with low HDL-C and 5.2% with high HDL-C, and all prioritized variants identified by NGS were confirmed by Sanger sequencing. Our results suggest that a molecular diagnosis can be identified in a substantial proportion of patients with low HDL-C using targeted NGS.

Keywords: ATP binding cassette transporter A1; atherosclerosis; diagnostic tools; genetics; genomics; high density lipoprotein; molecular diagnosis.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • ATP Binding Cassette Transporter 1 / blood
  • ATP Binding Cassette Transporter 1 / genetics*
  • Alleles
  • Cardiovascular Diseases / blood
  • Cardiovascular Diseases / diagnosis
  • Cardiovascular Diseases / genetics*
  • Cholesterol, HDL / blood*
  • Cholesterol, HDL / genetics*
  • Exons
  • Female
  • Genetic Association Studies
  • High-Throughput Nucleotide Sequencing / methods*
  • Humans
  • Hypercholesterolemia / blood*
  • Hypercholesterolemia / genetics*
  • Introns
  • Male
  • Middle Aged
  • Risk Factors

Substances

  • ABCA1 protein, human
  • ATP Binding Cassette Transporter 1
  • Cholesterol, HDL