Oxytocin improves behavioral and electrophysiological deficits in a novel Shank3-deficient rat

Elife. 2017 Jan 31:6:e18904. doi: 10.7554/eLife.18904.

Abstract

Mutations in the synaptic gene SHANK3 lead to a neurodevelopmental disorder known as Phelan-McDermid syndrome (PMS). PMS is a relatively common monogenic and highly penetrant cause of autism spectrum disorder (ASD) and intellectual disability (ID), and frequently presents with attention deficits. The underlying neurobiology of PMS is not fully known and pharmacological treatments for core symptoms do not exist. Here, we report the production and characterization of a Shank3-deficient rat model of PMS, with a genetic alteration similar to a human SHANK3 mutation. We show that Shank3-deficient rats exhibit impaired long-term social recognition memory and attention, and reduced synaptic plasticity in the hippocampal-medial prefrontal cortex pathway. These deficits were attenuated with oxytocin treatment. The effect of oxytocin on reversing non-social attention deficits is a particularly novel finding, and the results implicate an oxytocinergic contribution in this genetically defined subtype of ASD and ID, suggesting an individualized therapeutic approach for PMS.

Keywords: Phelan McDermid Syndrome; Shank3; attention; autism spectrum disorder; neuroscience; oxytocin; rat; social behavior.

Publication types

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

MeSH terms

  • Animals
  • Behavior, Animal / drug effects
  • Chromosome Deletion
  • Chromosome Disorders / drug therapy*
  • Chromosome Disorders / pathology
  • Chromosome Disorders / physiopathology
  • Chromosomes, Human, Pair 22
  • Disease Models, Animal
  • Hippocampus / pathology
  • Nerve Tissue Proteins / deficiency*
  • Oxytocin / administration & dosage*
  • Prefrontal Cortex / pathology
  • Rats
  • Social Behavior

Substances

  • Nerve Tissue Proteins
  • Shank3 protein, rat
  • Oxytocin

Supplementary concepts

  • Telomeric 22q13 Monosomy Syndrome