Molecular genetic testing and measurement of levels of GPIHBP1 autoantibodies in patients with severe hypertriglyceridemia: The importance of identifying the underlying cause of hypertriglyceridemia

J Clin Lipidol. 2024 Jan-Feb;18(1):e80-e89. doi: 10.1016/j.jacl.2023.11.002. Epub 2023 Nov 7.

Abstract

Background: Severe hypertriglyceridemia can be caused by pathogenic variants in genes encoding proteins involved in the metabolism of triglyceride-rich lipoproteins. A key protein in this respect is lipoprotein lipase (LPL) which hydrolyzes triglycerides in these lipoproteins. Another important protein is glycosylphosphatidylinositol-anchored high density lipoprotein-binding protein 1 (GPIHBP1) which transports LPL to the luminal side of the endothelial cells.

Objective: Our objective was to identify a genetic cause of hypertriglyceridemia in 459 consecutive unrelated subjects with levels of serum triglycerides ≥20 mmol/l. These patients had been referred for molecular genetic testing from 1998 to 2021. In addition, we wanted to study whether GPIHBP1 autoantibodies also were a cause of hypertriglyceridemia.

Methods: Molecular genetic analyses of the genes encoding LPL, GPIHBP1, apolipoprotein C2, lipase maturation factor 1 and apolipoprotein A5 as well as apolipoprotein E genotyping, were performed in all 459 patients. Serum was obtained from 132 of the patients for measurement of GPIHBP1 autoantibodies approximately nine years after molecular genetic testing was performed.

Results: A monogenic cause was found in four of the 459 (0.9%) patients, and nine (2.0%) patients had dyslipoproteinemia due to homozygosity for apolipoprotein E2. One of the 132 (0.8%) patients had GPIHBP1 autoantibody syndrome.

Conclusion: Only 0.9% of the patients had monogenic hypertriglyceridemia, and only 0.8% had GPIHBP1 autoantibody syndrome. The latter figure is most likely an underestimate because serum samples were obtained approximately nine years after hypertriglyceridemia was first identified. There is a need to implement measurement of GPIHBP1 autoantibodies in clinical medicine to secure that proper therapeutic actions are taken.

Keywords: Autoantibodies; Glycosylphosphatidylinositol-anchored high-density lipoprotein-binding protein 1; Lipoprotein lipase; Triglycerides; Variant.

MeSH terms

  • Apolipoproteins
  • Autoantibodies
  • Endothelial Cells
  • Humans
  • Hypertriglyceridemia* / genetics
  • Lipoprotein Lipase / genetics
  • Lipoprotein Lipase / metabolism
  • Lipoproteins
  • Molecular Biology
  • Receptors, Lipoprotein*
  • Triglycerides / metabolism

Substances

  • Autoantibodies
  • Receptors, Lipoprotein
  • Lipoprotein Lipase
  • Lipoproteins
  • Triglycerides
  • Apolipoproteins
  • GPIHBP1 protein, human