Mecp2 Deletion from Cholinergic Neurons Selectively Impairs Recognition Memory and Disrupts Cholinergic Modulation of the Perirhinal Cortex

eNeuro. 2019 Nov 1;6(6):ENEURO.0134-19.2019. doi: 10.1523/ENEURO.0134-19.2019. Print 2019 Nov/Dec.

Abstract

Rett Syndrome is a neurological disorder caused by mutations in the gene encoding methyl CpG binding protein 2 (MeCP2) and characterized by severe intellectual disability. The cholinergic system is a critical modulator of cognitive ability and is affected in patients with Rett Syndrome. To better understand the importance of MeCP2 function in cholinergic neurons, we studied the effect of selective Mecp2 deletion from cholinergic neurons in mice. Mice with Mecp2 deletion from cholinergic neurons were selectively impaired in assays of recognition memory, a cognitive task largely mediated by the perirhinal cortex (PRH). Deletion of Mecp2 from cholinergic neurons resulted in profound alterations in baseline firing of L5/6 neurons and eliminated the responses of these neurons to optogenetic stimulation of cholinergic input to PRH. Both the behavioral and the electrophysiological deficits of cholinergic Mecp2 deletion were rescued by inhibiting ACh breakdown with donepezil treatment.

Keywords: Mecp2; Rett Syndrome; acetylcholine; perirhinal; recognition.

Publication types

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

MeSH terms

  • Action Potentials / drug effects
  • Action Potentials / physiology
  • Animals
  • Cholinergic Neurons / drug effects
  • Cholinergic Neurons / metabolism*
  • Cholinesterase Inhibitors / pharmacology
  • Disease Models, Animal
  • Donepezil / pharmacology
  • Methyl-CpG-Binding Protein 2 / genetics
  • Methyl-CpG-Binding Protein 2 / metabolism*
  • Mice
  • Mice, Knockout
  • Optogenetics
  • Perirhinal Cortex / drug effects
  • Perirhinal Cortex / metabolism*
  • Phenotype
  • Recognition, Psychology / drug effects
  • Recognition, Psychology / physiology*
  • Rett Syndrome / genetics
  • Rett Syndrome / metabolism

Substances

  • Cholinesterase Inhibitors
  • Methyl-CpG-Binding Protein 2
  • Donepezil