Literature DB >> 21424692

Truncating mutations in NRXN2 and NRXN1 in autism spectrum disorders and schizophrenia.

Julie Gauthier1, Tabrez J Siddiqui, Peng Huashan, Daisaku Yokomaku, Fadi F Hamdan, Nathalie Champagne, Mathieu Lapointe, Dan Spiegelman, Anne Noreau, Ronald G Lafrenière, Ferid Fathalli, Ridha Joober, Marie-Odile Krebs, Lynn E DeLisi, Laurent Mottron, Eric Fombonne, Jacques L Michaud, Pierre Drapeau, Salvatore Carbonetto, Ann Marie Craig, Guy A Rouleau.   

Abstract

Growing genetic evidence is converging in favor of common pathogenic mechanisms for autism spectrum disorders (ASD), intellectual disability (ID or mental retardation) and schizophrenia (SCZ), three neurodevelopmental disorders affecting cognition and behavior. Copy number variations and deleterious mutations in synaptic organizing proteins including NRXN1 have been associated with these neurodevelopmental disorders, but no such associations have been reported for NRXN2 or NRXN3. From resequencing the three neurexin genes in individuals affected by ASD (n = 142), SCZ (n = 143) or non-syndromic ID (n = 94), we identified a truncating mutation in NRXN2 in a patient with ASD inherited from a father with severe language delay and family history of SCZ. We also identified a de novo truncating mutation in NRXN1 in a patient with SCZ, and other potential pathogenic ASD mutations. These truncating mutations result in proteins that fail to promote synaptic differentiation in neuron coculture and fail to bind either of the established postsynaptic binding partners LRRTM2 or NLGN2 in cell binding assays. Our findings link NRXN2 disruption to the pathogenesis of ASD for the first time and further strengthen the involvement of NRXN1 in SCZ, supporting the notion of a common genetic mechanism in these disorders.

Entities:  

Mesh:

Substances:

Year:  2011        PMID: 21424692      PMCID: PMC3204930          DOI: 10.1007/s00439-011-0975-z

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  44 in total

1.  CASK and protein 4.1 support F-actin nucleation on neurexins.

Authors:  T Biederer; T C Sudhof
Journal:  J Biol Chem       Date:  2001-10-16       Impact factor: 5.157

2.  Alpha-neurexins couple Ca2+ channels to synaptic vesicle exocytosis.

Authors:  Markus Missler; Weiqi Zhang; Astrid Rohlmann; Gunnar Kattenstroth; Robert E Hammer; Kurt Gottmann; Thomas C Südhof
Journal:  Nature       Date:  2003-06-26       Impact factor: 49.962

3.  Neurexins: synaptic cell surface proteins related to the alpha-latrotoxin receptor and laminin.

Authors:  Y A Ushkaryov; A G Petrenko; M Geppert; T C Südhof
Journal:  Science       Date:  1992-07-03       Impact factor: 47.728

Review 4.  Sensorimotor gating and schizophrenia. Human and animal model studies.

Authors:  D L Braff; M A Geyer
Journal:  Arch Gen Psychiatry       Date:  1990-02

Review 5.  Assessing the validity of an animal model of deficient sensorimotor gating in schizophrenic patients.

Authors:  N R Swerdlow; D L Braff; N Taaid; M A Geyer
Journal:  Arch Gen Psychiatry       Date:  1994-02

6.  X-linked mental retardation and autism are associated with a mutation in the NLGN4 gene, a member of the neuroligin family.

Authors:  Frédéric Laumonnier; Frédérique Bonnet-Brilhault; Marie Gomot; Romuald Blanc; Albert David; Marie-Pierre Moizard; Martine Raynaud; Nathalie Ronce; Eric Lemonnier; Patrick Calvas; Béatrice Laudier; Jamel Chelly; Jean-Pierre Fryns; Hans-Hilger Ropers; Ben C J Hamel; Christian Andres; Catherine Barthélémy; Claude Moraine; Sylvain Briault
Journal:  Am J Hum Genet       Date:  2004-02-12       Impact factor: 11.025

7.  Molecular characterisation of the 22q13 deletion syndrome supports the role of haploinsufficiency of SHANK3/PROSAP2 in the major neurological symptoms.

Authors:  H L Wilson; A C C Wong; S R Shaw; W-Y Tse; G A Stapleton; M C Phelan; S Hu; J Marshall; H E McDermid
Journal:  J Med Genet       Date:  2003-08       Impact factor: 6.318

8.  Neuroligin 1: a splice site-specific ligand for beta-neurexins.

Authors:  K Ichtchenko; Y Hata; T Nguyen; B Ullrich; M Missler; C Moomaw; T C Südhof
Journal:  Cell       Date:  1995-05-05       Impact factor: 41.582

Review 9.  Autism as a disorder of neural information processing: directions for research and targets for therapy.

Authors:  M K Belmonte; E H Cook; G M Anderson; J L R Rubenstein; W T Greenough; A Beckel-Mitchener; E Courchesne; L M Boulanger; S B Powell; P R Levitt; E K Perry; Y H Jiang; T M DeLorey; E Tierney
Journal:  Mol Psychiatry       Date:  2004-07       Impact factor: 15.992

10.  A stoichiometric complex of neurexins and dystroglycan in brain.

Authors:  S Sugita; F Saito; J Tang; J Satz; K Campbell; T C Südhof
Journal:  J Cell Biol       Date:  2001-07-23       Impact factor: 10.539

View more
  110 in total

1.  Human Neuropsychiatric Disease Modeling using Conditional Deletion Reveals Synaptic Transmission Defects Caused by Heterozygous Mutations in NRXN1.

Authors:  ChangHui Pak; Tamas Danko; Yingsha Zhang; Jason Aoto; Garret Anderson; Stephan Maxeiner; Fei Yi; Marius Wernig; Thomas C Südhof
Journal:  Cell Stem Cell       Date:  2015-08-13       Impact factor: 24.633

Review 2.  Autoantibodies to Synaptic Receptors and Neuronal Cell Surface Proteins in Autoimmune Diseases of the Central Nervous System.

Authors:  Josep Dalmau; Christian Geis; Francesc Graus
Journal:  Physiol Rev       Date:  2017-04       Impact factor: 37.312

3.  Developmental delays and reduced pup ultrasonic vocalizations but normal sociability in mice lacking the postsynaptic cell adhesion protein neuroligin2.

Authors:  Markus Wöhr; Jill L Silverman; Maria L Scattoni; Sarah M Turner; Mark J Harris; Roheeni Saxena; Jacqueline N Crawley
Journal:  Behav Brain Res       Date:  2012-07-20       Impact factor: 3.332

Review 4.  A matter of balance: role of neurexin and neuroligin at the synapse.

Authors:  Marie Louise Bang; Sylwia Owczarek
Journal:  Neurochem Res       Date:  2013-04-05       Impact factor: 3.996

5.  Structural Plasticity of Neurexin 1α: Implications for its Role as Synaptic Organizer.

Authors:  Jianfang Liu; Anurag Misra; M V V V Sekhar Reddy; Mark Andrew White; Gang Ren; Gabby Rudenko
Journal:  J Mol Biol       Date:  2018-09-05       Impact factor: 5.469

6.  Impaired hippocampal neuroligin-2 function by chronic stress or synthetic peptide treatment is linked to social deficits and increased aggression.

Authors:  Michael A van der Kooij; Martina Fantin; Igor Kraev; Irina Korshunova; Jocelyn Grosse; Olivia Zanoletti; Ramon Guirado; Clara Garcia-Mompó; Juan Nacher; Michael G Stewart; Vladimir Berezin; Carmen Sandi
Journal:  Neuropsychopharmacology       Date:  2013-11-11       Impact factor: 7.853

7.  The specific α-neurexin interactor calsyntenin-3 promotes excitatory and inhibitory synapse development.

Authors:  Katherine L Pettem; Daisaku Yokomaku; Lin Luo; Michael W Linhoff; Tuhina Prasad; Steven A Connor; Tabrez J Siddiqui; Hiroshi Kawabe; Fang Chen; Ling Zhang; Gabby Rudenko; Yu Tian Wang; Nils Brose; Ann Marie Craig
Journal:  Neuron       Date:  2013-10-02       Impact factor: 17.173

8.  A rare exonic NRXN3 deletion segregating with neurodevelopmental and neuropsychiatric conditions in a three-generation Chinese family.

Authors:  Haiming Yuan; Qingming Wang; Yanhui Liu; Wei Yang; Yi He; James F Gusella; Jiage Song; Yiping Shen
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2018-08-04       Impact factor: 3.568

Review 9.  The genetics of Autism Spectrum Disorders--a guide for clinicians.

Authors:  Karsten M Heil; Christian P Schaaf
Journal:  Curr Psychiatry Rep       Date:  2013-01       Impact factor: 5.285

10.  Adeno-associated viral overexpression of neuroligin 2 in the mouse hippocampus enhances GABAergic synapses and impairs hippocampal-dependent behaviors.

Authors:  M Van Zandt; E Weiss; A Almyasheva; S Lipior; S Maisel; J R Naegele
Journal:  Behav Brain Res       Date:  2018-12-31       Impact factor: 3.332

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.