Compromising the phosphodependent regulation of the GABA A R β3 subunit reproduces the core phenotypes of autism spectrum disorders

Abstract

Alterations in the efficacy of neuronal inhibition mediated by GABA A receptors (GABA A Rs) containing β3 subunits are continually implicated in autism spectrum disorders (ASDs). In vitro, the plasma membrane stability of GABA A Rs is potentiated via phosphorylation of serine residues 408 and 409 (S408/9) in the β3 subunit, an effect that is mimicked by their mutation to alanines. Here, we created a mouse in which S408/9 have been mutated to alanines (S408/9A). S408/9A mice exhibited altered dendritic spine structure, increased repetitive behavior, decreased social interaction, and an epileptic phenotype. Thus, mutation of S408/9 reproduces the core deficits seen in humans with ASDs. Collectively, our results suggest that alterations in phosphorylation and/or activity of β3-containing GABA A Rs may directly contribute to the pathophysiology of ASDs.

Document Details

Document Type
Pub Defense Publication
Publication Date
Nov 16, 2015
Source ID
10.1073/pnas.1514657112

Entities

People

  • Amit Modgil
  • Armen M. Abramian
  • Jamie Maguire
  • Joshua Walker
  • Miho Terunuma
  • Nicholas J. Brandon
  • Paul A. Davies
  • Rachel Jurd
  • Stephen J. Moss
  • Thuy N. Vien
  • Uwe Rudolph

Organizations

  • AstraZeneca
  • Congressionally Directed Medical Research Programs
  • Harvard Medical School
  • National Institute of Mental Health
  • National Institute of Neurological Disorders and Stroke
  • Simons Foundation
  • Tufts University
  • University College London
  • University of Leicester

Tags

Fields of Study

  • Biology

Readers

  • Molecular Biology and Genetics
  • Neuroscience
  • Traumatic Brain Injury (TBI) and Cognitive Aging in the Guam and Border Populations Affected by Alzheimer's Disease and Tau-Associated Dementias.