Plasma Glucosylsphingosine in GBA1 Mutation Carriers with and without Parkinson's Disease

Mov Disord. 2022 Feb;37(2):416-421. doi: 10.1002/mds.28846. Epub 2021 Nov 6.

Abstract

Background: Biallelic mutations in the GBA1 gene encoding glucocerebrosidase cause Gaucher's disease, whereas heterozygous carriers are at risk for Parkinson's disease (PD). Glucosylsphingosine is a clinically meaningful biomarker of Gaucher's disease but could not be assayed previously in heterozygous GBA1 carriers.

Objective: The aim of this study was to assess plasma glucosylsphingosine levels in GBA1 N370S carriers with and without PD.

Methods: Glucosylsphingosine, glucosylceramide, and four other lipids were quantified in plasma from N370S heterozygotes with (n = 20) or without (n = 20) PD, healthy controls (n = 20), idiopathic PD (n = 20), and four N370S homozygotes (positive controls; Gaucher's/PD) using quantitative ultra-performance liquid chromatography tandem mass spectrometry.

Results: Plasma glucosylsphingosine was significantly higher in N370S heterozygotes compared with noncarriers, independent of disease status. As expected, Gaucher's/PD cases showed increases in both glucocerebrosidase substrates, glucosylsphingosine and glucosylceramide.

Conclusions: Plasma glucosylsphingosine accumulation in N370S heterozygotes shown in this study opens up its future assessment as a clinically meaningful biomarker of GBA1-PD. © 2021 International Parkinson and Movement Disorder Society.

Keywords: Gaucher's; Parkinson's; glucocerebrosidase; lipidomics.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Gaucher Disease* / genetics
  • Glucosylceramidase / genetics
  • Humans
  • Mutation / genetics
  • Parkinson Disease* / genetics
  • Psychosine / analogs & derivatives

Substances

  • Psychosine
  • sphingosyl beta-glucoside
  • GBA protein, human
  • Glucosylceramidase