Bioenergetic Defects and Oxidative Damage in Transgenic Mouse Models of Neurodegenerative Disorders

Abstract

This project aims to determine the roles of bioenergetic dysfunction and oxidative stress in mechanisms of neurodegeneration in Huntington's disease (HD) and familial amyotrophic lateral sclerosis. Studies in this second year extended the first year's investigations into cerebral glucose metabolism in mutant mouse models of HE). We found increases in cerebral glucose use in forebrain regions of 4 month-old Hdh "knock-in" mice expressing a mutant 92 glutamine stretch in huntingtin protein (Hdh to the Q92), relative to levels in wild-type mice (Hdh to the Q7). Metabolic changes precede pathologic changes and show a gene-dosage effect, homozygote Hdh to the Q92 mice showing larger magnitude changes than heterozygotes. We attempted to measure similar parameters in R6/2 HD mice, but experiments were hindered by the diabetic profile of these mice. However, findings in another HE) mouse model (N171-82Q) also show pre-symptomatic cerebral glucose use elevations associated with the gene mutation. Results from years 1 and 2 suggest that mutant huntingtin expression induces metabolic abnormalities that precede symptom onset and pathological events in these mouse HE) models. Further, the occurrence of metabolic changes is dependent on polyglutamine repeat length and gene dosage. These findings represent significant progress towards the original goals of this proposal.

Open PDF

Document Details

Document Type
Technical Report
Publication Date
Jun 01, 2001
Accession Number
ADA397702

Entities

People

  • Susan Browne

Organizations

  • Cornell University

Tags

DTIC Thesaurus Topics

  • Amino Acids
  • Brain
  • Cell Physiological Processes
  • Cells
  • Cellular Structures
  • Chemistry
  • Medical Personnel
  • Metabolic Diseases
  • Movement Disorders
  • Neurodegeneration
  • Neurons
  • Neurosciences

Fields of Study

  • Biology

Readers

  • Molecular and Cellular Biology
  • Molecular and genetic basis of cancer.
  • Neuroscience