Literature DB >> 22539775

Negative allosteric modulation of the mGluR5 receptor reduces repetitive behaviors and rescues social deficits in mouse models of autism.

Jill L Silverman1, Daniel G Smith, Stacey J Sukoff Rizzo, Michael N Karras, Sarah M Turner, Seda S Tolu, Dianne K Bryce, Deborah L Smith, Kari Fonseca, Robert H Ring, Jacqueline N Crawley.   

Abstract

Neurodevelopmental disorders such as autism and fragile X syndrome were long thought to be medically untreatable, on the assumption that brain dysfunctions were immutably hardwired before diagnosis. Recent revelations that many cases of autism are caused by mutations in genes that control the ongoing formation and maturation of synapses have challenged this dogma. Antagonists of metabotropic glutamate receptor subtype 5 (mGluR5), which modulate excitatory neurotransmission, are in clinical trials for fragile X syndrome, a major genetic cause of intellectual disabilities. About 30% of patients with fragile X syndrome meet the diagnostic criteria for autism. Reasoning by analogy, we considered the mGluR5 receptor as a potential target for intervention in autism. We used BTBR T+tf/J (BTBR) mice, an established model with robust behavioral phenotypes relevant to the three diagnostic behavioral symptoms of autism--unusual social interactions, impaired communication, and repetitive behaviors--to probe the efficacy of a selective negative allosteric modulator of the mGluR5 receptor, GRN-529. GRN-529 reduced repetitive behaviors in three cohorts of BTBR mice at doses that did not induce sedation in control assays of open field locomotion. In addition, the same nonsedating doses reduced the spontaneous stereotyped jumping that characterizes a second inbred strain of mice, C58/J. Further, GRN-529 partially reversed the striking lack of sociability in BTBR mice on some parameters of social approach and reciprocal social interactions. These findings raise the possibility that a single targeted pharmacological intervention may alleviate multiple diagnostic behavioral symptoms of autism.

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Year:  2012        PMID: 22539775      PMCID: PMC4904784          DOI: 10.1126/scitranslmed.3003501

Source DB:  PubMed          Journal:  Sci Transl Med        ISSN: 1946-6234            Impact factor:   17.956


  97 in total

1.  Reduced scent marking and ultrasonic vocalizations in the BTBR T+tf/J mouse model of autism.

Authors:  M Wöhr; F I Roullet; J N Crawley
Journal:  Genes Brain Behav       Date:  2011-02       Impact factor: 3.449

2.  The autism diagnostic observation schedule-generic: a standard measure of social and communication deficits associated with the spectrum of autism.

Authors:  C Lord; S Risi; L Lambrecht; E H Cook; B L Leventhal; P C DiLavore; A Pickles; M Rutter
Journal:  J Autism Dev Disord       Date:  2000-06

3.  Deconstructing sociability, an autism-relevant phenotype, in mouse models.

Authors:  Andrew H Fairless; Rhia Y Shah; Ashley J Guthrie; Hongzhe Li; Edward S Brodkin
Journal:  Anat Rec (Hoboken)       Date:  2011-09-08       Impact factor: 2.064

Review 4.  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

5.  Autism genome-wide copy number variation reveals ubiquitin and neuronal genes.

Authors:  Joseph T Glessner; Kai Wang; Guiqing Cai; Olena Korvatska; Cecilia E Kim; Shawn Wood; Haitao Zhang; Annette Estes; Camille W Brune; Jonathan P Bradfield; Marcin Imielinski; Edward C Frackelton; Jennifer Reichert; Emily L Crawford; Jeffrey Munson; Patrick M A Sleiman; Rosetta Chiavacci; Kiran Annaiah; Kelly Thomas; Cuiping Hou; Wendy Glaberson; James Flory; Frederick Otieno; Maria Garris; Latha Soorya; Lambertus Klei; Joseph Piven; Kacie J Meyer; Evdokia Anagnostou; Takeshi Sakurai; Rachel M Game; Danielle S Rudd; Danielle Zurawiecki; Christopher J McDougle; Lea K Davis; Judith Miller; David J Posey; Shana Michaels; Alexander Kolevzon; Jeremy M Silverman; Raphael Bernier; Susan E Levy; Robert T Schultz; Geraldine Dawson; Thomas Owley; William M McMahon; Thomas H Wassink; John A Sweeney; John I Nurnberger; Hilary Coon; James S Sutcliffe; Nancy J Minshew; Struan F A Grant; Maja Bucan; Edwin H Cook; Joseph D Buxbaum; Bernie Devlin; Gerard D Schellenberg; Hakon Hakonarson
Journal:  Nature       Date:  2009-04-28       Impact factor: 49.962

6.  Haploinsufficiency for Pten and Serotonin transporter cooperatively influences brain size and social behavior.

Authors:  Damon T Page; Orsolya J Kuti; Chrysa Prestia; Mriganka Sur
Journal:  Proc Natl Acad Sci U S A       Date:  2009-02-10       Impact factor: 11.205

7.  Postnatal lesion evidence against a primary role for the corpus callosum in mouse sociability.

Authors:  Mu Yang; Andrew M Clarke; Jacqueline N Crawley
Journal:  Eur J Neurosci       Date:  2009-04       Impact factor: 3.386

Review 8.  Diagnosis of autism spectrum disorders in the first 3 years of life.

Authors:  Rebecca J Landa
Journal:  Nat Clin Pract Neurol       Date:  2008-02-05

Review 9.  Autism spectrum disorders--a genetics review.

Authors:  Judith H Miles
Journal:  Genet Med       Date:  2011-04       Impact factor: 8.822

10.  Shank3 mutant mice display autistic-like behaviours and striatal dysfunction.

Authors:  João Peça; Cátia Feliciano; Jonathan T Ting; Wenting Wang; Michael F Wells; Talaignair N Venkatraman; Christopher D Lascola; Zhanyan Fu; Guoping Feng
Journal:  Nature       Date:  2011-03-20       Impact factor: 49.962

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  116 in total

1.  Drugs targeting mGluR5 receptor offer 'fragile' hope for autism.

Authors:  Sarah C P Williams
Journal:  Nat Med       Date:  2012-06-06       Impact factor: 53.440

2.  Neurodevelopmental disorders: Glutamate blockers show benefit in models of autism spectrum disorders.

Authors:  Charlotte Harrison
Journal:  Nat Rev Drug Discov       Date:  2012-06-01       Impact factor: 84.694

3.  Insulin-Like Growth Factor II Targets the mTOR Pathway to Reverse Autism-Like Phenotypes in Mice.

Authors:  Adam B Steinmetz; Sarah A Stern; Amy S Kohtz; Giannina Descalzi; Cristina M Alberini
Journal:  J Neurosci       Date:  2017-12-07       Impact factor: 6.167

Review 4.  Advancing the discovery of medications for autism spectrum disorder using new technologies to reveal social brain circuitry in rodents.

Authors:  Martien J Kas; Meera E Modi; Michael D Saxe; Daniel G Smith
Journal:  Psychopharmacology (Berl)       Date:  2014-02-13       Impact factor: 4.530

5.  Parvalbumin cell ablation of NMDA-R1 causes increased resting network excitability with associated social and self-care deficits.

Authors:  Eddie N Billingslea; Valerie M Tatard-Leitman; Jaynie Anguiano; Catherine R Jutzeler; Jimmy Suh; John A Saunders; Susumu Morita; Robert E Featherstone; Pavel I Ortinski; Michael J Gandal; Robert Lin; Yuling Liang; Raquel E Gur; Gregory C Carlson; Chang-Gyu Hahn; Steven J Siegel
Journal:  Neuropsychopharmacology       Date:  2014-02-14       Impact factor: 7.853

6.  Repetitive behavior profile and supersensitivity to amphetamine in the C58/J mouse model of autism.

Authors:  Sheryl S Moy; Natallia V Riddick; Viktoriya D Nikolova; Brian L Teng; Kara L Agster; Randal J Nonneman; Nancy B Young; Lorinda K Baker; Jessica J Nadler; James W Bodfish
Journal:  Behav Brain Res       Date:  2013-11-08       Impact factor: 3.332

7.  Acetylcholine elevation relieves cognitive rigidity and social deficiency in a mouse model of autism.

Authors:  Golan Karvat; Tali Kimchi
Journal:  Neuropsychopharmacology       Date:  2013-10-07       Impact factor: 7.853

Review 8.  Approaches to Understanding Multisensory Dysfunction in Autism Spectrum Disorder.

Authors:  Justin K Siemann; Jeremy Veenstra-VanderWeele; Mark T Wallace
Journal:  Autism Res       Date:  2020-09-01       Impact factor: 5.216

Review 9.  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

Review 10.  Oxytocin and vasopressin systems in genetic syndromes and neurodevelopmental disorders.

Authors:  S M Francis; A Sagar; T Levin-Decanini; W Liu; C S Carter; S Jacob
Journal:  Brain Res       Date:  2014-01-22       Impact factor: 3.252

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