Literature DB >> 21733672

Synaptic microcircuit dysfunction in genetic models of neurodevelopmental disorders: focus on Mecp2 and Met.

Gordon M G Shepherd1, David M Katz.   

Abstract

Recent findings in the genetics of neurodevelopmental syndromes have ushered in an exciting era of discovery in which substrates of neurologic dysfunction are being identified at the synaptic and microcircuit levels in mouse models of these disorders. We review recent progress in this area, focusing on two examples of mouse models of autism spectrum disorders (ASDs): Mecp2 models of Rett syndrome, and a Met-knockout model of non-syndromic forms of autism. In both cases, a dominant theme is changes in synaptic strength, associated with hyper-connectivity or hypo-connectivity in specific microcircuits. Alterations in intrinsic neuronal excitability are also found, but do not appear to be as common. The microcircuit-specific nature of synaptic changes observed in these ASD models indicates that it will be necessary to define mechanisms of circuit dysfunction on a case-by-case basis, not only in neocortex but also in brainstem and other sub-cortical areas. Thus, functional microcircuit analysis is emerging as an important line of investigation, highly complementary to neurogenetic and molecular strategies, and holds promise for generating models of the underlying pathophysiology and for guiding development of novel therapeutic strategies.
Copyright © 2011 Elsevier Ltd. All rights reserved.

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Year:  2011        PMID: 21733672      PMCID: PMC3199024          DOI: 10.1016/j.conb.2011.06.006

Source DB:  PubMed          Journal:  Curr Opin Neurobiol        ISSN: 0959-4388            Impact factor:   6.627


  80 in total

Review 1.  The developmental neurobiology of autism spectrum disorder.

Authors:  Emanuel DiCicco-Bloom; Catherine Lord; Lonnie Zwaigenbaum; Eric Courchesne; Stephen R Dager; Christoph Schmitz; Robert T Schultz; Jacqueline Crawley; Larry J Young
Journal:  J Neurosci       Date:  2006-06-28       Impact factor: 6.167

Review 2.  Neurogeometry and potential synaptic connectivity.

Authors:  Armen Stepanyants; Dmitri B Chklovskii
Journal:  Trends Neurosci       Date:  2005-07       Impact factor: 13.837

3.  Dendritic spine pathologies in hippocampal pyramidal neurons from Rett syndrome brain and after expression of Rett-associated MECP2 mutations.

Authors:  Christopher A Chapleau; Gaston D Calfa; Meredith C Lane; Asher J Albertson; Jennifer L Larimore; Shinichi Kudo; Dawna L Armstrong; Alan K Percy; Lucas Pozzo-Miller
Journal:  Neurobiol Dis       Date:  2009-05-12       Impact factor: 5.996

4.  Loss of silent-chromatin looping and impaired imprinting of DLX5 in Rett syndrome.

Authors:  Shin-ichi Horike; Shutao Cai; Masaru Miyano; Jan-Fang Cheng; Terumi Kohwi-Shigematsu
Journal:  Nat Genet       Date:  2004-12-19       Impact factor: 38.330

5.  Partial reversal of Rett Syndrome-like symptoms in MeCP2 mutant mice.

Authors:  Daniela Tropea; Emanuela Giacometti; Nathan R Wilson; Caroline Beard; Cortina McCurry; Dong Dong Fu; Ruth Flannery; Rudolf Jaenisch; Mriganka Sur
Journal:  Proc Natl Acad Sci U S A       Date:  2009-02-10       Impact factor: 11.205

6.  Synaptic circuit abnormalities of motor-frontal layer 2/3 pyramidal neurons in an RNA interference model of methyl-CpG-binding protein 2 deficiency.

Authors:  Lydia Wood; Noah W Gray; Zhaolan Zhou; Michael E Greenberg; Gordon M G Shepherd
Journal:  J Neurosci       Date:  2009-10-07       Impact factor: 6.167

7.  Mecp2 deficiency leads to delayed maturation and altered gene expression in hippocampal neurons.

Authors:  Richard D Smrt; Julialea Eaves-Egenes; Basam Z Barkho; Nicholas J Santistevan; Chunmei Zhao; James B Aimone; Fred H Gage; Xinyu Zhao
Journal:  Neurobiol Dis       Date:  2007-04-27       Impact factor: 5.996

8.  Disruption of cerebral cortex MET signaling in autism spectrum disorder.

Authors:  Daniel B Campbell; Rosanna D'Oronzio; Krassi Garbett; Philip J Ebert; Karoly Mirnics; Pat Levitt; Antonio M Persico
Journal:  Ann Neurol       Date:  2007-09       Impact factor: 10.422

9.  Early breathing defects after moderate hypoxia or hypercapnia in a mouse model of Rett syndrome.

Authors:  Nicolas Voituron; Sébastien Zanella; Clément Menuet; Mathias Dutschmann; Gérard Hilaire
Journal:  Respir Physiol Neurobiol       Date:  2009-06-11       Impact factor: 1.931

10.  Dysfunction in GABA signalling mediates autism-like stereotypies and Rett syndrome phenotypes.

Authors:  Hsiao-Tuan Chao; Hongmei Chen; Rodney C Samaco; Mingshan Xue; Maria Chahrour; Jong Yoo; Jeffrey L Neul; Shiaoching Gong; Hui-Chen Lu; Nathaniel Heintz; Marc Ekker; John L R Rubenstein; Jeffrey L Noebels; Christian Rosenmund; Huda Y Zoghbi
Journal:  Nature       Date:  2010-11-11       Impact factor: 49.962

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  42 in total

1.  Mechanisms of Functional Hypoconnectivity in the Medial Prefrontal Cortex of Mecp2 Null Mice.

Authors:  Michael P Sceniak; Min Lang; Addison C Enomoto; C James Howell; Douglas J Hermes; David M Katz
Journal:  Cereb Cortex       Date:  2015-02-07       Impact factor: 5.357

2.  Emergence of functional subnetworks in layer 2/3 cortex induced by sequential spikes in vivo.

Authors:  Taekeun Kim; Won Chan Oh; Joon Ho Choi; Hyung-Bae Kwon
Journal:  Proc Natl Acad Sci U S A       Date:  2016-02-22       Impact factor: 11.205

3.  NEXMIF/KIDLIA Knock-out Mouse Demonstrates Autism-Like Behaviors, Memory Deficits, and Impairments in Synapse Formation and Function.

Authors:  James Gilbert; Margaret O'Connor; Sebastian Templet; Mahsa Moghaddam; Anaïs Di Via Ioschpe; Amanda Sinclair; Ling-Qiang Zhu; Weifeng Xu; Heng-Ye Man
Journal:  J Neurosci       Date:  2019-11-08       Impact factor: 6.167

Review 4.  Circadian dysfunction may be a key component of the non-motor symptoms of Parkinson's disease: insights from a transgenic mouse model.

Authors:  L David Willison; Takashi Kudo; Dawn H Loh; Dika Kuljis; Christopher S Colwell
Journal:  Exp Neurol       Date:  2013-01-24       Impact factor: 5.330

5.  Brain activity mapping in Mecp2 mutant mice reveals functional deficits in forebrain circuits, including key nodes in the default mode network, that are reversed with ketamine treatment.

Authors:  Miriam Kron; C James Howell; Ian T Adams; Michael Ransbottom; Diana Christian; Michael Ogier; David M Katz
Journal:  J Neurosci       Date:  2012-10-03       Impact factor: 6.167

6.  NMDA receptor regulation prevents regression of visual cortical function in the absence of Mecp2.

Authors:  Severine Durand; Annarita Patrizi; Kathleen B Quast; Lea Hachigian; Roman Pavlyuk; Alka Saxena; Piero Carninci; Takao K Hensch; Michela Fagiolini
Journal:  Neuron       Date:  2012-12-20       Impact factor: 17.173

Review 7.  Therapeutic approaches for shankopathies.

Authors:  Xiaoming Wang; Alexandra L Bey; Leeyup Chung; Andrew D Krystal; Yong-Hui Jiang
Journal:  Dev Neurobiol       Date:  2013-10-11       Impact factor: 3.964

8.  Neurobiology meets genomic science: the promise of human-induced pluripotent stem cells.

Authors:  Hanna E Stevens; Jessica Mariani; Gianfilippo Coppola; Flora M Vaccarino
Journal:  Dev Psychopathol       Date:  2012-11

9.  Loss of function of KIAA2022 causes mild to severe intellectual disability with an autism spectrum disorder and impairs neurite outgrowth.

Authors:  Lionel Van Maldergem; Qingming Hou; Vera M Kalscheuer; Marlène Rio; Martine Doco-Fenzy; Ana Medeira; Arjan P M de Brouwer; Christelle Cabrol; Stefan A Haas; Pierre Cacciagli; Sébastien Moutton; Emilie Landais; Jacques Motte; Laurence Colleaux; Céline Bonnet; Laurent Villard; Juliette Dupont; Heng-Ye Man
Journal:  Hum Mol Genet       Date:  2013-04-24       Impact factor: 6.150

10.  MeCP2 regulates ethanol sensitivity and intake.

Authors:  Vez Repunte-Canonigo; Jihuan Chen; Celine Lefebvre; Tomoya Kawamura; Max Kreifeldt; Oan Basson; Amanda J Roberts; Pietro Paolo Sanna
Journal:  Addict Biol       Date:  2013-02-28       Impact factor: 4.280

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