Early correction of synaptic long-term depression improves abnormal anxiety-like behavior in adult GluN2B-C456Y-mutant mice

PLoS Biol. 2020 Apr 30;18(4):e3000717. doi: 10.1371/journal.pbio.3000717. eCollection 2020 Apr.

Abstract

Extensive evidence links Glutamate receptor, ionotropic, NMDA2B (GRIN2B), encoding the GluN2B/NR2B subunit of N-methyl-D-aspartate receptors (NMDARs), with various neurodevelopmental disorders, including autism spectrum disorders (ASDs), but the underlying mechanisms remain unclear. In addition, it remains unknown whether mutations in GluN2B, which starts to be expressed early in development, induces early pathophysiology that can be corrected by early treatments for long-lasting effects. We generated and characterized Grin2b-mutant mice that carry a heterozygous, ASD-risk C456Y mutation (Grin2b+/C456Y). In Grin2b+/C456Y mice, GluN2B protein levels were strongly reduced in association with decreased hippocampal NMDAR currents and NMDAR-dependent long-term depression (LTD) but unaltered long-term potentiation, indicative of mutation-induced protein degradation and LTD sensitivity. Behaviorally, Grin2b+/C456Y mice showed normal social interaction but exhibited abnormal anxiolytic-like behavior. Importantly, early, but not late, treatment of young Grin2b+/C456Y mice with the NMDAR agonist D-cycloserine rescued NMDAR currents and LTD in juvenile mice and improved anxiolytic-like behavior in adult mice. Therefore, GluN2B-C456Y haploinsufficiency decreases GluN2B protein levels, NMDAR-dependent LTD, and anxiety-like behavior, and early activation of NMDAR function has long-lasting effects on adult mouse behavior.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Anxiety / genetics*
  • Anxiety / physiopathology
  • Behavior, Animal / drug effects
  • Cycloserine / pharmacology
  • Excitatory Postsynaptic Potentials / genetics
  • Gene Knock-In Techniques
  • Haploinsufficiency / genetics
  • Heterozygote
  • Hippocampus / metabolism
  • Hippocampus / physiology*
  • Long-Term Synaptic Depression / drug effects
  • Long-Term Synaptic Depression / physiology*
  • Mice, Mutant Strains
  • Mutation
  • Nerve Tissue Proteins / metabolism
  • Receptors, N-Methyl-D-Aspartate / agonists
  • Receptors, N-Methyl-D-Aspartate / genetics*
  • Receptors, N-Methyl-D-Aspartate / metabolism

Substances

  • Gprin1 protein, mouse
  • NR2B NMDA receptor
  • Nerve Tissue Proteins
  • Receptors, N-Methyl-D-Aspartate
  • Cycloserine

Grants and funding

This work was supported by the European Research Council (ERC Advanced Grant #693021 to PP), the University Pierre-et-Marie-Curie (UPMC Paris 6) and the Fondation pour la Recherche Médicale (FRM) (fellowships to BS), the National Research Foundation of Korea (NRF) grant funded by the Korea government (MSIT, NRF-2017R1A5A2015391 to YCB), and the Institute for Basic Science (IBS-R002-D1 to EK). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.