Glutaric aciduria I: creatine supplementation restores creatinephosphate levels in mixed cortex cells from rat incubated with 3-hydroxyglutarate

Mol Genet Metab. 2003 Feb;78(2):108-11. doi: 10.1016/s1096-7192(02)00227-5.

Abstract

The pathogenesis of neurological sequelae in glutaric aciduria I (GA I) is still unclear. Some evidence exists for compromised energy generation in the brain of patients with GA I resulting in 'slow-onset' excitotoxicity. Previously, we have shown a reduced activity of the mitochondrial ATPsynthase in cultured mixed cortex cells from neonatal rats incubated with 2-4mM 3-hydroxyglutarate (3-OH glut) for 24h. In the present study we measured cellular contents of high energy phosphate compounds (creatinephosphate CP, ATP, and ADP) in this model after a 24h incubation period with 2-4mM glutarate (glut) or 3-OH glut. 3-OH glut specifically led to a reduction of CP content in a dose-dependent manner, whereas concentrations of ATP, ADP, and AMP remained unchanged. The drop in CP-concentration could be prevented by preincubation with the non-competitive NMDA-receptor antagonist MK 801 or coincubation with 1mM creatine. NMDA-receptor associated ion channels may be opened due to a lack of energy inside the neurons caused by a reduction of CP. This is followed by membrane depolarization which could impair electrogenic creatine transport into the cell.

MeSH terms

  • Adenine Nucleotides / metabolism
  • Animals
  • Animals, Newborn
  • Cerebral Cortex / cytology
  • Cerebral Cortex / metabolism*
  • Creatine / administration & dosage
  • Creatine / therapeutic use*
  • Glutarates / administration & dosage*
  • Glutarates / urine*
  • Metabolism, Inborn Errors / drug therapy*
  • Metabolism, Inborn Errors / metabolism
  • Phosphocreatine / metabolism*
  • Rats

Substances

  • 3-hydroxyglutaric acid
  • Adenine Nucleotides
  • Glutarates
  • Phosphocreatine
  • glutaric acid
  • Creatine