Gene network disruptions and neurogenesis defects in the adult Ts1Cje mouse model of Down syndrome

PLoS One. 2010 Jul 16;5(7):e11561. doi: 10.1371/journal.pone.0011561.

Abstract

Background: Down syndrome (DS) individuals suffer mental retardation with further cognitive decline and early onset Alzheimer's disease.

Methodology/principal findings: To understand how trisomy 21 causes these neurological abnormalities we investigated changes in gene expression networks combined with a systematic cell lineage analysis of adult neurogenesis using the Ts1Cje mouse model of DS. We demonstrated down regulation of a number of key genes involved in proliferation and cell cycle progression including Mcm7, Brca2, Prim1, Cenpo and Aurka in trisomic neurospheres. We found that trisomy did not affect the number of adult neural stem cells but resulted in reduced numbers of neural progenitors and neuroblasts. Analysis of differentiating adult Ts1Cje neural progenitors showed a severe reduction in numbers of neurons produced with a tendency for less elaborate neurites, whilst the numbers of astrocytes was increased.

Conclusions/significance: We have shown that trisomy affects a number of elements of adult neurogenesis likely to result in a progressive pathogenesis and consequently providing the potential for the development of therapies to slow progression of, or even ameliorate the neuronal deficits suffered by DS individuals.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Apoptosis / genetics
  • Apoptosis / physiology
  • Aurora Kinase A
  • Aurora Kinases
  • BRCA2 Protein / genetics
  • Cell Cycle Proteins / genetics
  • Cell Differentiation / genetics
  • Cell Differentiation / physiology
  • Cell Movement / genetics
  • Cell Movement / physiology
  • Cells, Cultured
  • DNA-Binding Proteins / genetics
  • Disease Models, Animal
  • Down Syndrome / genetics
  • Down Syndrome / metabolism*
  • Down Syndrome / pathology*
  • Female
  • Fluorescent Antibody Technique
  • Gene Regulatory Networks / genetics*
  • Immunohistochemistry
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Minichromosome Maintenance Complex Component 7
  • Neurogenesis / genetics
  • Neurogenesis / physiology*
  • Neurons / cytology
  • Neurons / metabolism
  • Nuclear Proteins / genetics
  • Oligonucleotide Array Sequence Analysis
  • Protein Serine-Threonine Kinases / genetics
  • Reverse Transcriptase Polymerase Chain Reaction
  • Stem Cells / cytology
  • Stem Cells / metabolism
  • Trisomy / genetics

Substances

  • BRCA2 Protein
  • BRCA2 protein, mouse
  • Cell Cycle Proteins
  • DNA-Binding Proteins
  • Nuclear Proteins
  • Aurka protein, mouse
  • Aurora Kinase A
  • Aurora Kinases
  • Protein Serine-Threonine Kinases
  • Mcm7 protein, mouse
  • Minichromosome Maintenance Complex Component 7