In Vivo Chimeric Alzheimer's Disease Modeling of Apolipoprotein E4 Toxicity in Human Neurons

Cell Rep. 2020 Jul 28;32(4):107962. doi: 10.1016/j.celrep.2020.107962.

Abstract

Despite its clear impact on Alzheimer's disease (AD) risk, apolipoprotein (apo) E4's contributions to AD etiology remain poorly understood. Progress in answering this and other questions in AD research has been limited by an inability to model human-specific phenotypes in an in vivo environment. Here we transplant human induced pluripotent stem cell (hiPSC)-derived neurons carrying normal apoE3 or pathogenic apoE4 into human apoE3 or apoE4 knockin mouse hippocampi, enabling us to disentangle the effects of apoE4 produced in human neurons and in the brain environment. Using single-nucleus RNA sequencing (snRNA-seq), we identify key transcriptional changes specific to human neuron subtypes in response to endogenous or exogenous apoE4. We also find that Aβ from transplanted human neurons forms plaque-like aggregates, with differences in localization and interaction with microglia depending on the transplant and host apoE genotype. These findings highlight the power of in vivo chimeric disease modeling for studying AD.

Keywords: Alzheimer’s disease; Aβ; apoE4; chimeric disease modeling; excitatory neuron; human iPSC; inhibitory neuron; microglia; snRNA-seq; transcriptomics.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Alzheimer Disease / physiopathology*
  • Amyloid beta-Peptides / metabolism
  • Animals
  • Apolipoprotein E3 / genetics
  • Apolipoprotein E3 / metabolism
  • Apolipoprotein E3 / pharmacology
  • Apolipoprotein E4 / genetics
  • Apolipoprotein E4 / metabolism*
  • Apolipoproteins E / genetics
  • Apolipoproteins E / metabolism
  • Brain / metabolism
  • Chimera / genetics
  • Chimera / metabolism
  • Gene Knock-In Techniques
  • Hippocampus / metabolism
  • Humans
  • Induced Pluripotent Stem Cells / metabolism
  • Mice
  • Mice, Inbred C57BL
  • Mice, Transgenic
  • Microglia / metabolism
  • Models, Biological
  • Neurons / metabolism*
  • tau Proteins / metabolism

Substances

  • Amyloid beta-Peptides
  • Apolipoprotein E3
  • Apolipoprotein E4
  • Apolipoproteins E
  • tau Proteins