Development of a brain-penetrant G9a methylase inhibitor to target Alzheimer's disease-associated proteopathology

Nat Commun. 2025 May 7;16(1):4222. doi: 10.1038/s41467-025-59128-z.

Abstract

Current Aβ-targeting therapeutics for Alzheimer's disease (AD) only slow cognitive decline due to poor understanding of AD pathogenesis. Here we describe a mechanism of AD pathogenesis in which the histone methyltransferase G9a noncanonically regulates translation of hippocampal proteins associated with AD pathology. Correspondingly, we developed a brain-penetrant inhibitor of G9a, MS1262, which restored both age-related learning & memory and noncognitive functions in multiple AD mouse models. Further, comparison of AD pathology-correlated mouse proteomes with those of AD patients found G9a regulates pathological pathways that promote Aβ and neurofibrillary tangles. This mouse-to-human overlap of G9a regulated AD-associated pathologic proteins supports at the molecular level the efficacy of targeting G9a translational mechanism for treating AD patients. Additionally, MS1262 treatment reversed the AD-characteristic expression or phosphorylation of multiple clinically validated biomarkers of AD that have the potential to be used for early-stage AD diagnosis and companion diagnosis of individualized drug effects.

MeSH terms

  • Alzheimer Disease* / drug therapy
  • Alzheimer Disease* / genetics
  • Alzheimer Disease* / metabolism
  • Alzheimer Disease* / pathology
  • Amyloid beta-Peptides / metabolism
  • Animals
  • Brain* / drug effects
  • Brain* / metabolism
  • Brain* / pathology
  • Disease Models, Animal
  • Enzyme Inhibitors* / pharmacology
  • Female
  • Hippocampus / drug effects
  • Hippocampus / metabolism
  • Hippocampus / pathology
  • Histocompatibility Antigens / metabolism
  • Histone-Lysine N-Methyltransferase* / antagonists & inhibitors
  • Histone-Lysine N-Methyltransferase* / genetics
  • Histone-Lysine N-Methyltransferase* / metabolism
  • Humans
  • Male
  • Mice
  • Mice, Transgenic
  • Neurofibrillary Tangles / metabolism
  • Neurofibrillary Tangles / pathology
  • Proteome / metabolism

Substances

  • Histone-Lysine N-Methyltransferase
  • Enzyme Inhibitors
  • EHMT2 protein, human
  • Amyloid beta-Peptides
  • G9a protein, mouse
  • Proteome
  • Histocompatibility Antigens