Early defect of transforming growth factor β1 formation in Huntington's disease

J Cell Mol Med. 2011 Mar;15(3):555-71. doi: 10.1111/j.1582-4934.2010.01011.x.

Abstract

A defective expression or activity of neurotrophic factors, such as brain- and glial-derived neurotrophic factors, contributes to neuronal damage in Huntington's disease (HD). Here, we focused on transforming growth factor-β (TGF-β(1) ), a pleiotropic cytokine with an established role in mechanisms of neuroprotection. Asymptomatic HD patients showed a reduction in TGF-β(1) levels in the peripheral blood, which was related to trinucleotide mutation length and glucose hypometabolism in the caudate nucleus. Immunohistochemical analysis in post-mortem brain tissues showed that TGF-β(1) was reduced in cortical neurons of asymptomatic and symptomatic HD patients. Both YAC128 and R6/2 HD mutant mice showed a reduced expression of TGF-β(1) in the cerebral cortex, localized in neurons, but not in astrocytes. We examined the pharmacological regulation of TGF-β(1) formation in asymptomatic R6/2 mice, where blood TGF-β(1) levels were also reduced. In these R6/2 mice, both the mGlu2/3 metabotropic glutamate receptor agonist, LY379268, and riluzole failed to increase TGF-β(1) formation in the cerebral cortex and corpus striatum, suggesting that a defect in the regulation of TGF-β(1) production is associated with HD. Accordingly, reduced TGF-β(1) mRNA and protein levels were found in cultured astrocytes transfected with mutated exon 1 of the human huntingtin gene, and in striatal knock-in cell lines expressing full-length huntingtin with an expanded glutamine repeat. Taken together, our data suggest that serum TGF-β(1) levels are potential biomarkers of HD development during the asymptomatic phase of the disease, and raise the possibility that strategies aimed at rescuing TGF-β(1) levels in the brain may influence the progression of HD.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Amino Acids / pharmacology
  • Animals
  • Astrocytes / drug effects
  • Astrocytes / metabolism*
  • Blotting, Western
  • Brain / metabolism*
  • Brain / pathology
  • Bridged Bicyclo Compounds, Heterocyclic / pharmacology
  • Cells, Cultured
  • Female
  • Humans
  • Huntingtin Protein
  • Huntington Disease / blood
  • Huntington Disease / metabolism*
  • Huntington Disease / pathology
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Mice, Transgenic
  • Middle Aged
  • Mutation
  • Nerve Tissue Proteins / genetics
  • Nuclear Proteins / genetics
  • Positron-Emission Tomography
  • Postmortem Changes
  • Receptors, Metabotropic Glutamate / agonists
  • Reverse Transcriptase Polymerase Chain Reaction
  • Time Factors
  • Transforming Growth Factor beta1 / blood
  • Transforming Growth Factor beta1 / genetics
  • Transforming Growth Factor beta1 / metabolism*
  • Young Adult

Substances

  • Amino Acids
  • Bridged Bicyclo Compounds, Heterocyclic
  • HTT protein, human
  • Huntingtin Protein
  • LY 379268
  • Nerve Tissue Proteins
  • Nuclear Proteins
  • Receptors, Metabotropic Glutamate
  • Transforming Growth Factor beta1