Lentiviral delivery of human erythropoietin attenuates hippocampal atrophy and improves cognition in the R6/2 mouse model of Huntington's disease

Neurobiol Dis. 2020 Oct:144:105024. doi: 10.1016/j.nbd.2020.105024. Epub 2020 Jul 20.

Abstract

Huntington's disease (HD) is an incurable neurodegenerative disorder caused by a trinucleotide (CAG) repeat expansion in the huntingtin gene (HTT). The R6/2 transgenic mouse model of HD expresses exon 1 of the human HTT gene with approximately 150 CAG repeats. R6/2 mice develop progressive behavioural abnormalities, impaired neurogenesis, and atrophy of several brain regions. In recent years, erythropoietin (EPO) has been shown to confer neuroprotection and enhance neurogenesis, rendering it a promising molecule to attenuate HD symptoms. In this study, the therapeutic potential of EPO was evaluated in female R6/2 transgenic mice. A single bilateral injection of a lentivirus encoding human EPO (LV-hEPO) was performed into the lateral ventricles of R6/2 mice at disease onset (8 weeks of age). Control groups were either untreated or injected with a lentivirus encoding green fluorescent protein (LV-GFP). Thirty days after virus administration, hEPO mRNA and protein were present in injected R6/2 brains. Compared to control R6/2 mice, LV-hEPO-treated R6/2 mice exhibited reduced hippocampal atrophy, increased neuroblast branching towards the dentate granular cell layer, and improved spatial cognition. Our results suggest that LV-hEPO administration may be a promising strategy to reduce cognitive impairment in HD.

Keywords: Gene delivery; Huntington's disease; Intracerebroventricular; Neurodegeneration; Therapy; Transgenic mice.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Atrophy
  • Cognition*
  • Disease Models, Animal
  • Erythropoietin / genetics*
  • Erythropoietin / metabolism
  • Female
  • Genetic Therapy
  • Hippocampus / pathology*
  • Huntington Disease / pathology
  • Huntington Disease / physiopathology*
  • Injections, Intraventricular
  • Lentivirus
  • Mice
  • Mice, Transgenic
  • Neural Stem Cells
  • Organ Size
  • Spatial Navigation*
  • Transfection

Substances

  • EPO protein, human
  • Erythropoietin