Genetic load determines atrophy in hand cortico-striatal pathways in presymptomatic Huntington's disease

Hum Brain Mapp. 2018 Oct;39(10):3871-3883. doi: 10.1002/hbm.24217. Epub 2018 May 24.

Abstract

Huntington's disease (HD) is an inherited neurodegenerative disorder that causes progressive breakdown of striatal neurons. Standard white matter integrity measures like fractional anisotropy and mean diffusivity derived from diffusion tensor imaging were analyzed in prodromal-HD subjects; however, they studied either a whole brain or specific subcortical white matter structures with connections to cortical motor areas. In this work, we propose a novel analysis of a longitudinal cohort of 243 prodromal-HD individuals and 88 healthy controls who underwent two or more diffusion MRI scans as part of the PREDICT-HD study. We separately trace specific white matter fiber tracts connecting the striatum (caudate and putamen) with four cortical regions corresponding to the hand, face, trunk, and leg motor areas. A multi-tensor tractography algorithm with an isotropic volume fraction compartment allows estimating diffusion of fast-moving extra-cellular water in regions containing crossing fibers and provides quantification of a microstructural property related to tissue atrophy. The tissue atrophy rate is separately analyzed in eight cortico-striatal pathways as a function of CAG-repeats (genetic load) by statistically regressing out age effect from our cohort. The results demonstrate a statistically significant increase in isotropic volume fraction (atrophy) bilaterally in hand fiber connections to the putamen with increasing CAG-repeats, which connects the genetic abnormality (CAG-repeats) to an imaging-based microstructural marker of tissue integrity in specific white matter pathways in HD. Isotropic volume fraction measures in eight cortico-striatal pathways are also correlated significantly with total motor scores and diagnostic confidence levels, providing evidence of their relevance to HD clinical presentation.

Keywords: CAG-repeats; cortico-striatal pathways; diffusion MRI; isotropic volume fraction; prodromal-HD.

Publication types

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

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Atrophy / pathology
  • Caudate Nucleus / diagnostic imaging
  • Caudate Nucleus / pathology*
  • Diffusion Tensor Imaging / methods*
  • Female
  • Genetic Load*
  • Humans
  • Huntington Disease / diagnostic imaging
  • Huntington Disease / genetics*
  • Huntington Disease / pathology*
  • Longitudinal Studies
  • Male
  • Middle Aged
  • Motor Cortex / diagnostic imaging
  • Motor Cortex / pathology*
  • Neural Pathways / diagnostic imaging
  • Neural Pathways / pathology
  • Prodromal Symptoms*
  • Putamen / diagnostic imaging
  • Putamen / pathology*
  • Trinucleotide Repeats / genetics*
  • White Matter / diagnostic imaging
  • White Matter / pathology*
  • Young Adult