Literature DB >> 24999357

Convergent synaptic and circuit substrates underlying autism genetic risks.

Aaron McGee1, Guohui Li2, Zhongming Lu3, Shenfeng Qiu4.   

Abstract

There has been a surge of diagnosis of autism spectrum disorders (ASD) over the past decade. While large, high powered genome screening studies of children with ASD have identified numerous genetic risk factors, research efforts to understanding how each of these risk factors contributes to the development autism has met with limited success. Revealing the mechanisms by which these genetic risk factors affect brain development and predispose a child to autism requires mechanistic understanding of the neurobiological changes underlying this devastating group of developmental disorders at multifaceted molecular, cellular and system levels. It has been increasingly clear that the normal trajectory of neurodevelopment is compromised in autism, in multiple domains as much as aberrant neuronal production, growth, functional maturation, patterned connectivity, and balanced excitation and inhibition of brain networks. Many autism risk factors identified in humans have been now reconstituted in experimental mouse models to allow mechanistic interrogation of the biological role of the risk gene. Studies utilizing these mouse models have revealed that underlying the enormous heterogeneity of perturbed cellular events, mechanisms directing synaptic and circuit assembly may provide a unifying explanation for the pathophysiological changes and behavioral endophenotypes seen in autism, although synaptic perturbations are far from being the only alterations relevant for ASD. In this review, we discuss synaptic and circuit abnormalities obtained from several prevalent mouse models, particularly those reflecting syndromic forms of ASD that are caused by single gene perturbations. These compiled results reveal that ASD risk genes contribute to proper signaling of the developing gene networks that maintain synaptic and circuit homeostasis, which is fundamental to normal brain development.

Entities:  

Keywords:  autism spectrum disorders; behavior; circuits; development; neurodevelopmental disorders; risk genes; synapse

Year:  2014        PMID: 24999357      PMCID: PMC4079081          DOI: 10.1007/s11515-014-1298-y

Source DB:  PubMed          Journal:  Front Biol (Beijing)        ISSN: 1674-7984


  134 in total

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2.  Functional and anatomical cortical underconnectivity in autism: evidence from an FMRI study of an executive function task and corpus callosum morphometry.

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3.  Dendritic spine instability and insensitivity to modulation by sensory experience in a mouse model of fragile X syndrome.

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4.  Recessive symptomatic focal epilepsy and mutant contactin-associated protein-like 2.

Authors:  Kevin A Strauss; Erik G Puffenberger; Matthew J Huentelman; Steven Gottlieb; Seth E Dobrin; Jennifer M Parod; Dietrich A Stephan; D Holmes Morton
Journal:  N Engl J Med       Date:  2006-03-30       Impact factor: 91.245

Review 5.  Behavioural phenotyping assays for mouse models of autism.

Authors:  Jill L Silverman; Mu Yang; Catherine Lord; Jacqueline N Crawley
Journal:  Nat Rev Neurosci       Date:  2010-07       Impact factor: 34.870

Review 6.  Imprinting in Angelman and Prader-Willi syndromes.

Authors:  Y Jiang; T F Tsai; J Bressler; A L Beaudet
Journal:  Curr Opin Genet Dev       Date:  1998-06       Impact factor: 5.578

7.  Disruption of neurexin 1 associated with autism spectrum disorder.

Authors:  Hyung-Goo Kim; Shotaro Kishikawa; Anne W Higgins; Ihn-Sik Seong; Diana J Donovan; Yiping Shen; Eric Lally; Lauren A Weiss; Juliane Najm; Kerstin Kutsche; Maria Descartes; Lynn Holt; Stephen Braddock; Robin Troxell; Lee Kaplan; Fred Volkmar; Ami Klin; Katherine Tsatsanis; David J Harris; Ilse Noens; David L Pauls; Mark J Daly; Marcy E MacDonald; Cynthia C Morton; Bradley J Quade; James F Gusella
Journal:  Am J Hum Genet       Date:  2008-01       Impact factor: 11.025

8.  Rett syndrome as a minicolumnopathy.

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10.  Adult neural function requires MeCP2.

Authors:  Christopher M McGraw; Rodney C Samaco; Huda Y Zoghbi
Journal:  Science       Date:  2011-06-02       Impact factor: 47.728

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Journal:  Sci Transl Med       Date:  2015-05-06       Impact factor: 17.956

2.  The Autism Protein Ube3A/E6AP Remodels Neuronal Dendritic Arborization via Caspase-Dependent Microtubule Destabilization.

Authors:  Natasha Khatri; James P Gilbert; Yuda Huo; Roozhin Sharaflari; Michael Nee; Hui Qiao; Heng-Ye Man
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3.  The influence of NRXN1 on systemizing and the brain structure in healthy adults.

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Review 4.  A Subset of Autism-Associated Genes Regulate the Structural Stability of Neurons.

Authors:  Yu-Chih Lin; Jeannine A Frei; Michaela B C Kilander; Wenjuan Shen; Gene J Blatt
Journal:  Front Cell Neurosci       Date:  2016-11-17       Impact factor: 5.505

Review 5.  Common Ribs of Inhibitory Synaptic Dysfunction in the Umbrella of Neurodevelopmental Disorders.

Authors:  Rachel Ali Rodriguez; Christina Joya; Rochelle M Hines
Journal:  Front Mol Neurosci       Date:  2018-04-24       Impact factor: 5.639

Review 6.  Autism Spectrum Disorder and miRNA: An Overview of Experimental Models.

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Journal:  Brain Sci       Date:  2019-10-03
  6 in total

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