CAG repeats determine brain atrophy in spinocerebellar ataxia 17: a VBM study

PLoS One. 2011 Jan 19;6(1):e15125. doi: 10.1371/journal.pone.0015125.

Abstract

Background: Abnormal repeat length has been associated with an earlier age of onset and more severe disease progression in the rare neurodegenerative disorder spinocerebellar ataxia 17 (SCA17).

Methodology/principal findings: To determine whether specific structural brain degeneration and rate of disease progression in SCA17 might be associated with the CAG repeat size, observer-independent voxel-based morphometry was applied to high-resolution magnetic resonance images of 16 patients with SCA17 and 16 age-matched healthy controls. The main finding contrasting SCA17 patients with healthy controls demonstrated atrophy in the cerebellum bilaterally. Multiple regression analyses with available genetic data and also post-hoc correlations revealed an inverse relationship again with cerebellar atrophy. Moreover, we found an inverse relationship between the CAG repeat length and rate of disease progression.

Conclusions: Our results highlight the fundamental role of the cerebellum in this neurodegenerative disease and support the genotype-phenotype relationship in SCA17 patients. Genetic factors may determine individual susceptibility to neurodegeneration and rate of disease progression.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Age of Onset
  • Atrophy / genetics
  • Atrophy / metabolism
  • Brain / pathology*
  • Case-Control Studies
  • Cerebellum / pathology
  • Disease Progression
  • Female
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Nerve Degeneration / genetics
  • Spinocerebellar Ataxias / genetics*
  • Spinocerebellar Ataxias / pathology*
  • Trinucleotide Repeats*