Protective variant for hippocampal atrophy identified by whole exome sequencing

Ann Neurol. 2015 Mar;77(3):547-52. doi: 10.1002/ana.24349. Epub 2015 Feb 14.

Abstract

We used whole-exome sequencing to identify variants other than APOE associated with the rate of hippocampal atrophy in amnestic mild cognitive impairment. An in-silico predicted missense variant in REST (rs3796529) was found exclusively in subjects with slow hippocampal volume loss and validated using unbiased whole-brain analysis and meta-analysis across 5 independent cohorts. REST is a master regulator of neurogenesis and neuronal differentiation that has not been previously implicated in Alzheimer's disease. These findings nominate REST and its functional pathways as protective and illustrate the potential of combining next-generation sequencing with neuroimaging to discover novel disease mechanisms and potential therapeutic targets.

Publication types

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

MeSH terms

  • Aged
  • Alzheimer Disease / genetics
  • Alzheimer Disease / pathology
  • Alzheimer Disease / physiopathology
  • Amnesia / genetics*
  • Amnesia / pathology
  • Amnesia / physiopathology
  • Atrophy / genetics
  • Atrophy / pathology
  • Cognitive Dysfunction / genetics*
  • Cognitive Dysfunction / pathology
  • Cognitive Dysfunction / physiopathology
  • Disease Progression*
  • Exome / genetics*
  • Hippocampus / pathology*
  • Hippocampus / physiopathology
  • Humans
  • Male
  • Mutation, Missense
  • Protective Factors
  • Repressor Proteins / genetics*
  • Sequence Analysis, DNA / methods

Substances

  • RE1-silencing transcription factor
  • Repressor Proteins