Repeat-Associated Non-ATG Translation: Molecular Mechanisms and Contribution to Neurological Disease

Annu Rev Neurosci. 2019 Jul 8:42:227-247. doi: 10.1146/annurev-neuro-070918-050405. Epub 2019 Mar 25.

Abstract

Microsatellite mutations involving the expansion of tri-, tetra-, penta-, or hexanucleotide repeats cause more than 40 different neurological disorders. Although, traditionally, the position of the repeat within or outside of an open reading frame has been used to focus research on disease mechanisms involving protein loss of function, protein gain of function, or RNA gain of function, the discoveries of bidirectional transcription and repeat-associated non-ATG (RAN) have blurred these distinctions. Here we review what is known about RAN proteins in disease, the mechanisms by which they are produced, and the novel therapeutic opportunities they provide.

Keywords: RAN translation; mechanism; microsatellite repeat diseases; therapy; toxicity; translation.

Publication types

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

MeSH terms

  • Codon, Initiator / genetics
  • DNA Repeat Expansion / genetics*
  • Endoplasmic Reticulum Stress
  • Eukaryotic Initiation Factor-2 / physiology
  • Gain of Function Mutation
  • Genetic Code
  • Humans
  • Loss of Function Mutation
  • Microsatellite Repeats / genetics
  • Nerve Tissue Proteins / genetics*
  • Nervous System Diseases / genetics*
  • Neurodegenerative Diseases / genetics
  • Neurodegenerative Diseases / metabolism
  • Protein Biosynthesis*
  • Transcription, Genetic

Substances

  • Codon, Initiator
  • Eukaryotic Initiation Factor-2
  • Nerve Tissue Proteins