Cell type-specific over-expression of chromosome 21 genes in fibroblasts and fetal hearts with trisomy 21

BMC Med Genet. 2006 Mar 15:7:24. doi: 10.1186/1471-2350-7-24.

Abstract

Background: Down syndrome (DS) is caused by trisomy 21 (+21), but the aberrations in gene expression resulting from this chromosomal aneuploidy are not yet completely understood.

Methods: We used oligonucleotide microarrays to survey mRNA expression in early- and late-passage control and +21 fibroblasts and mid-gestation fetal hearts. We supplemented this analysis with northern blotting, western blotting, real-time RT-PCR, and immunohistochemistry.

Results: We found chromosome 21 genes consistently over-represented among the genes over-expressed in the +21 samples. However, these sets of over-expressed genes differed across the three cell/tissue types. The chromosome 21 gene MX1 was strongly over-expressed (mean 16-fold) in senescent +21 fibroblasts, a result verified by northern and western blotting. MX1 is an interferon target gene, and its mRNA was induced by interferons present in +21 fibroblast conditioned medium, suggesting an autocrine loop for its over-expression. By immunohistochemistry the p78MX1 protein was induced in lesional tissue of alopecia areata, an autoimmune disorder associated with DS. We found strong over-expression of the purine biosynthesis gene GART (mean 3-fold) in fetal hearts with +21 and verified this result by northern blotting and real-time RT-PCR.

Conclusion: Different subsets of chromosome 21 genes are over-expressed in different cell types with +21, and for some genes this over-expression is non-linear (>1.5X). Hyperactive interferon signaling is a candidate pathway for cell senescence and autoimmune disorders in DS, and abnormal purine metabolism should be investigated for a potential role in cardiac defects.

Publication types

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

MeSH terms

  • Alopecia Areata / genetics
  • Alopecia Areata / metabolism
  • Alopecia Areata / pathology
  • Carbon-Nitrogen Ligases / genetics
  • Carbon-Nitrogen Ligases / metabolism
  • Cells, Cultured
  • Chromosomes, Human, Pair 21*
  • Down Syndrome / embryology
  • Down Syndrome / genetics*
  • Down Syndrome / metabolism
  • Fetal Heart / metabolism*
  • Fibroblasts / metabolism*
  • GTP-Binding Proteins / biosynthesis
  • GTP-Binding Proteins / genetics
  • Gene Expression
  • Gene Expression Profiling
  • Humans
  • Multienzyme Complexes / genetics
  • Multienzyme Complexes / metabolism
  • Myxovirus Resistance Proteins
  • Phosphoribosylglycinamide Formyltransferase / genetics
  • Phosphoribosylglycinamide Formyltransferase / metabolism

Substances

  • Multienzyme Complexes
  • Myxovirus Resistance Proteins
  • Phosphoribosylglycinamide Formyltransferase
  • GTP-Binding Proteins
  • Carbon-Nitrogen Ligases
  • GART protein, human