Combined beta-glucosylceramide and ambroxol hydrochloride in patients with Gaucher related Parkinson disease: From clinical observations to drug development

Blood Cells Mol Dis. 2018 Feb:68:117-120. doi: 10.1016/j.bcmd.2016.10.028. Epub 2016 Nov 12.

Abstract

Both patients with non-neuronopathic Gaucher disease (GD) and heterozygous GBA mutation carrier are at increased risk for Parkinson disease (PD). The risk for PD in these groups does not linearly increase with glucosylceramide (GC) accumulation or with acid β-glucocerebrosidase (GCase) activity. This observation, together with other clinical systemic observations raises the possibility that extra-cellular GC actually has beneficial, anti-inflammatory, properties. Based on this hypothesis, we suggest here that the administration of supplementary oral GC to GBA carriers at risk for PD may slow inflammatory-driven secondary neuronal death. Such a treatment may act synergistically in GBA carriers once given in combination with an agent that prevent the primary pathologic process that leads to cell death. Ambroxol hydrochloride, a pharmacological chaperone, which reduces endoplasmic reticulum (ER) stress induced by accumulation of mutant misfolded GCase could serve as such an agent. The efficacy of this combined therapy, derived from clinical observations, in vivo and in vitro studies, should be evaluated in clinical trials.

Keywords: Ambroxol hydrochloride; Beta glucosylceramide; Gaucher disease; Parkinson disease.

Publication types

  • Review
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Ambroxol / therapeutic use*
  • Animals
  • Anti-Inflammatory Agents / therapeutic use*
  • Drug Discovery
  • Expectorants / therapeutic use*
  • Gaucher Disease / complications*
  • Gaucher Disease / drug therapy*
  • Gaucher Disease / genetics
  • Gaucher Disease / pathology
  • Glucosylceramidase / genetics
  • Glucosylceramides / therapeutic use*
  • Humans
  • Mutation
  • Parkinson Disease / drug therapy*
  • Parkinson Disease / etiology*
  • Parkinson Disease / genetics
  • Parkinson Disease / pathology

Substances

  • Anti-Inflammatory Agents
  • Expectorants
  • Glucosylceramides
  • Ambroxol
  • Glucosylceramidase