Literature DB >> 35181828

The GluN2B-Trp373 NMDA Receptor Variant is Associated with Autism-, Epilepsy-Related Phenotypes and Reduces NMDA Receptor Currents in Rats.

Xiaona Wang1, Zhiyue Guo2, Daoqi Mei3, Yaodong Zhang4, Shuai Zhao4, Shunan Hu4, Shuying Luo4, Qi Wang5, Chao Gao6.   

Abstract

Autism spectrum disorder (ASD) is a neurodevelopmental condition with core clinical features of abnormal communication, social interactions, atypical intelligence, and a higher risk of epilepsy. Prior work has suggested that de novo heterozygous mutations in the GRIN2B gene that encodes the GluN2B subunit of N-methyl-D-aspartic acid receptors are likely linked to ASD. However, whether GLuN2B-Trp373 mutation derived from autistic individuals causes ASD-like behavioral aberrations in rats remains to be determined. Here, through in utero electroporation and in vivo studies, we conducted a battery of tests to examine ASD-associated behaviors, cognitive impairments, and susceptibility to pentylenetetrazol-induced seizures. Whole-cell patch recording was utilized to determine whether the GluN2B-Trp373 mutation influences GluN2B-containing NMDA receptor currents in rats. Results show that, behaviorally, GLuN2B-Trp373 mutant rats exhibited core behavioral manifestations of ASD, such as social interaction deficits, increases in stereotyped behaviors and anxiety stereotyped/repetitive, impaired spatial memory, and enhanced risk of pentylenetetrazol-induced seizures, consistent with many of the hallmarks of low-functioning ASD in humans. Functionally, the GluN2B-Trp373 mutation results in reduced GluN2B surface protein expression together with decreased hippocampal NMDA receptor currents. Collectively, our findings highlight that GluN2B-Trp373 mutations can drive the manifestation of ASD-associated symptoms via the suppression of NMDA receptor currents.
© 2022. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  Autism; Epilepsy; GluN2B; Human diseases; NMDA receptors

Mesh:

Substances:

Year:  2022        PMID: 35181828     DOI: 10.1007/s11064-022-03554-8

Source DB:  PubMed          Journal:  Neurochem Res        ISSN: 0364-3190            Impact factor:   3.996


  47 in total

Review 1.  Epilepsy and autism: is there a special relationship?

Authors:  Anne T Berg; Sigita Plioplys
Journal:  Epilepsy Behav       Date:  2012-02-29       Impact factor: 2.937

Review 2.  Autism Spectrum Disorder and Epilepsy: Two Sides of the Same Coin?

Authors:  Shafali Spurling Jeste; Roberto Tuchman
Journal:  J Child Neurol       Date:  2015-09-14       Impact factor: 1.987

3.  Distinct Pathogenic Genes Causing Intellectual Disability and Autism Exhibit a Common Neuronal Network Hyperactivity Phenotype.

Authors:  Monica Frega; Martijn Selten; Britt Mossink; Jason M Keller; Katrin Linda; Rebecca Moerschen; Jieqiong Qu; Pierre Koerner; Sophie Jansen; Astrid Oudakker; Tjitske Kleefstra; Hans van Bokhoven; Huiqing Zhou; Dirk Schubert; Nael Nadif Kasri
Journal:  Cell Rep       Date:  2020-01-07       Impact factor: 9.423

4.  Missense Variants in the Histone Acetyltransferase Complex Component Gene TRRAP Cause Autism and Syndromic Intellectual Disability.

Authors:  Benjamin Cogné; Sophie Ehresmann; Eliane Beauregard-Lacroix; Justine Rousseau; Thomas Besnard; Thomas Garcia; Slavé Petrovski; Shiri Avni; Kirsty McWalter; Patrick R Blackburn; Stephan J Sanders; Kévin Uguen; Jacqueline Harris; Julie S Cohen; Moira Blyth; Anna Lehman; Jonathan Berg; Mindy H Li; Usha Kini; Shelagh Joss; Charlotte von der Lippe; Christopher T Gordon; Jennifer B Humberson; Laurie Robak; Daryl A Scott; Vernon R Sutton; Cara M Skraban; Jennifer J Johnston; Annapurna Poduri; Magnus Nordenskjöld; Vandana Shashi; Erica H Gerkes; Ernie M H F Bongers; Christian Gilissen; Yuri A Zarate; Malin Kvarnung; Kevin P Lally; Peggy A Kulch; Brina Daniels; Andres Hernandez-Garcia; Nicholas Stong; Julie McGaughran; Kyle Retterer; Kristian Tveten; Jennifer Sullivan; Madeleine R Geisheker; Asbjorg Stray-Pedersen; Jennifer M Tarpinian; Eric W Klee; Julie C Sapp; Jacob Zyskind; Øystein L Holla; Emma Bedoukian; Francesca Filippini; Anne Guimier; Arnaud Picard; Øyvind L Busk; Jaya Punetha; Rolph Pfundt; Anna Lindstrand; Ann Nordgren; Fayth Kalb; Megha Desai; Ashley Harmon Ebanks; Shalini N Jhangiani; Tammie Dewan; Zeynep H Coban Akdemir; Aida Telegrafi; Elaine H Zackai; Amber Begtrup; Xiaofei Song; Annick Toutain; Ingrid M Wentzensen; Sylvie Odent; Dominique Bonneau; Xénia Latypova; Wallid Deb; Sylvia Redon; Frédéric Bilan; Marine Legendre; Caitlin Troyer; Kerri Whitlock; Oana Caluseriu; Marine I Murphree; Pavel N Pichurin; Katherine Agre; Ralitza Gavrilova; Tuula Rinne; Meredith Park; Catherine Shain; Erin L Heinzen; Rui Xiao; Jeanne Amiel; Stanislas Lyonnet; Bertrand Isidor; Leslie G Biesecker; Dan Lowenstein; Jennifer E Posey; Anne-Sophie Denommé-Pichon; Claude Férec; Xiang-Jiao Yang; Jill A Rosenfeld; Brigitte Gilbert-Dussardier; Séverine Audebert-Bellanger; Richard Redon; Holly A F Stessman; Christoffer Nellaker; Yaping Yang; James R Lupski; David B Goldstein; Evan E Eichler; Francois Bolduc; Stéphane Bézieau; Sébastien Küry; Philippe M Campeau
Journal:  Am J Hum Genet       Date:  2019-02-28       Impact factor: 11.025

Review 5.  A de novo convergence of autism genetics and molecular neuroscience.

Authors:  Niklas Krumm; Brian J O'Roak; Jay Shendure; Evan E Eichler
Journal:  Trends Neurosci       Date:  2013-12-30       Impact factor: 13.837

6.  Synaptic, transcriptional and chromatin genes disrupted in autism.

Authors:  Silvia De Rubeis; Xin He; Arthur P Goldberg; Christopher S Poultney; Kaitlin Samocha; A Erucment Cicek; Yan Kou; Li Liu; Menachem Fromer; Susan Walker; Tarinder Singh; Lambertus Klei; Jack Kosmicki; Fu Shih-Chen; Branko Aleksic; Monica Biscaldi; Patrick F Bolton; Jessica M Brownfeld; Jinlu Cai; Nicholas G Campbell; Angel Carracedo; Maria H Chahrour; Andreas G Chiocchetti; Hilary Coon; Emily L Crawford; Sarah R Curran; Geraldine Dawson; Eftichia Duketis; Bridget A Fernandez; Louise Gallagher; Evan Geller; Stephen J Guter; R Sean Hill; Juliana Ionita-Laza; Patricia Jimenz Gonzalez; Helena Kilpinen; Sabine M Klauck; Alexander Kolevzon; Irene Lee; Irene Lei; Jing Lei; Terho Lehtimäki; Chiao-Feng Lin; Avi Ma'ayan; Christian R Marshall; Alison L McInnes; Benjamin Neale; Michael J Owen; Noriio Ozaki; Mara Parellada; Jeremy R Parr; Shaun Purcell; Kaija Puura; Deepthi Rajagopalan; Karola Rehnström; Abraham Reichenberg; Aniko Sabo; Michael Sachse; Stephan J Sanders; Chad Schafer; Martin Schulte-Rüther; David Skuse; Christine Stevens; Peter Szatmari; Kristiina Tammimies; Otto Valladares; Annette Voran; Wang Li-San; Lauren A Weiss; A Jeremy Willsey; Timothy W Yu; Ryan K C Yuen; Edwin H Cook; Christine M Freitag; Michael Gill; Christina M Hultman; Thomas Lehner; Aaarno Palotie; Gerard D Schellenberg; Pamela Sklar; Matthew W State; James S Sutcliffe; Christiopher A Walsh; Stephen W Scherer; Michael E Zwick; Jeffrey C Barett; David J Cutler; Kathryn Roeder; Bernie Devlin; Mark J Daly; Joseph D Buxbaum
Journal:  Nature       Date:  2014-10-29       Impact factor: 49.962

7.  Neuroligin 2 regulates absence seizures and behavioral arrests through GABAergic transmission within the thalamocortical circuitry.

Authors:  Feng Cao; Jackie J Liu; Susan Zhou; Miguel A Cortez; O Carter Snead; Jing Han; Zhengping Jia
Journal:  Nat Commun       Date:  2020-07-27       Impact factor: 14.919

8.  Patterns of de novo tandem repeat mutations and their role in autism.

Authors:  Ileena Mitra; Bonnie Huang; Nima Mousavi; Nichole Ma; Michael Lamkin; Richard Yanicky; Sharona Shleizer-Burko; Kirk E Lohmueller; Melissa Gymrek
Journal:  Nature       Date:  2021-01-13       Impact factor: 69.504

9.  Targeted sequencing identifies 91 neurodevelopmental-disorder risk genes with autism and developmental-disability biases.

Authors:  Holly A F Stessman; Bo Xiong; Bradley P Coe; Tianyun Wang; Kendra Hoekzema; Michaela Fenckova; Malin Kvarnung; Jennifer Gerdts; Sandy Trinh; Nele Cosemans; Laura Vives; Janice Lin; Tychele N Turner; Gijs Santen; Claudia Ruivenkamp; Marjolein Kriek; Arie van Haeringen; Emmelien Aten; Kathryn Friend; Jan Liebelt; Christopher Barnett; Eric Haan; Marie Shaw; Jozef Gecz; Britt-Marie Anderlid; Ann Nordgren; Anna Lindstrand; Charles Schwartz; R Frank Kooy; Geert Vandeweyer; Celine Helsmoortel; Corrado Romano; Antonino Alberti; Mirella Vinci; Emanuela Avola; Stefania Giusto; Eric Courchesne; Tiziano Pramparo; Karen Pierce; Srinivasa Nalabolu; David G Amaral; Ingrid E Scheffer; Martin B Delatycki; Paul J Lockhart; Fereydoun Hormozdiari; Benjamin Harich; Anna Castells-Nobau; Kun Xia; Hilde Peeters; Magnus Nordenskjöld; Annette Schenck; Raphael A Bernier; Evan E Eichler
Journal:  Nat Genet       Date:  2017-02-13       Impact factor: 38.330

10.  Endoplasmic reticulum retention and degradation of a mutation in SLC6A1 associated with epilepsy and autism.

Authors:  Jie Wang; Sarah Poliquin; Felicia Mermer; Jaclyn Eissman; Eric Delpire; Juexin Wang; Wangzhen Shen; Kefu Cai; Bing-Mei Li; Zong-Yan Li; Dong Xu; Gerald Nwosu; Carson Flamm; Wei-Ping Liao; Yi-Wu Shi; Jing-Qiong Kang
Journal:  Mol Brain       Date:  2020-05-12       Impact factor: 4.041

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