H63D mutation in hemochromatosis alters cholesterol metabolism and induces memory impairment

Neurobiol Aging. 2014 Jun;35(6):1511.e1-12. doi: 10.1016/j.neurobiolaging.2013.12.014. Epub 2013 Dec 25.

Abstract

The H63D variant of the hemochromatosis (HFE) gene, when expressed in carriers of the apolipoprotein E4 allele, is implicated as a risk factor for earlier onset of Alzheimer's disease (AD). We tested the hypothesis that like expression of apolipoprotein E4, expression of H63D-HFE disrupts cholesterol metabolism contributing to an increase in neurodegeneration and memory deficits. Analysis of SH-SY5Y human neuroblastoma cells transfected to stably express either wild type- (WT) or H63D-HFE indicated about a 50% reduction in cholesterol content in cells expressing H63D-HFE. This was accompanied by a significant decrease in expression of 3-hydroxy-3-methyl-glutaryl-CoA reductase, and a significant increase in expression of cholesterol 24-hydroxylase. Consistent with these studies, H67D-HFE (orthologous to human H63D-HFE) knock-in mice, showed a greater age dependent decline in brain cholesterol than WT-HFE animals and changes in expression of proteins regulating cholesterol metabolism. Brains of aged H67D-HFE mice also exhibited a significant decrease in expression of synapse proteins and a significant increase in caspase-3 expression relative to WT-HFE controls. H67D-HFE mice also had a greater reduction in brain volume and poorer recognition and spatial memory than WT-HFE mice, symptoms associated with AD. These results indicate that the alterations in cholesterol metabolism associated with expression of H63D-HFE may contribute to the development of AD.

Keywords: Alzheimer disease; Cholesterol; Hemochromatosis gene; Memory; Neurodegeneration.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Alleles
  • Alzheimer Disease / genetics
  • Alzheimer Disease / pathology
  • Alzheimer Disease / psychology
  • Animals
  • Apolipoprotein E4 / genetics
  • Brain / metabolism
  • Brain / pathology
  • Caspase 3 / metabolism
  • Cholesterol / metabolism*
  • Cholesterol 24-Hydroxylase
  • Gene Expression
  • Hemochromatosis / genetics*
  • Hemochromatosis / metabolism
  • Hemochromatosis Protein
  • Heterozygote
  • Histocompatibility Antigens Class I
  • Humans
  • Membrane Proteins / genetics*
  • Memory
  • Memory Disorders / genetics*
  • Memory Disorders / pathology
  • Memory Disorders / psychology
  • Mice
  • Mice, Transgenic
  • Mutation / genetics*
  • Neurodegenerative Diseases / genetics
  • Neurodegenerative Diseases / pathology
  • Neurodegenerative Diseases / psychology
  • Recognition, Psychology
  • Risk Factors
  • Steroid Hydroxylases / metabolism
  • Tumor Cells, Cultured

Substances

  • Apolipoprotein E4
  • HFE protein, human
  • Hemochromatosis Protein
  • Histocompatibility Antigens Class I
  • Membrane Proteins
  • Cholesterol
  • Steroid Hydroxylases
  • Cholesterol 24-Hydroxylase
  • Caspase 3