Reduced social interaction and ultrasonic communication in a mouse model of monogenic heritable autism

被引:399
作者
Jamain, Stephane [1 ,3 ,4 ]
Radyushkin, Konstantin
Hammerschmidt, Kurt [5 ]
Granon, Sylvie [2 ]
Boretius, Susann [6 ]
Varoqueaux, Frederique [3 ,4 ]
Ramanantsoa, Nelina [7 ]
Gallego, Jorge [7 ]
Ronnenberg, Anja
Winter, Dorina
Frahm, Jens
Fischer, Julia [5 ]
Bourgeron, Thomas [1 ,8 ]
Ehrenreich, Hannelore
Brose, Nils [3 ,4 ]
机构
[1] Inst Pasteur, Dept Neurosci, Grp Genet Humaine & Fonct Cognit, F-75724 Paris, France
[2] Inst Pasteur, Dept Neurosci, Unite Neurobiol Integrat Syst Cholinerg, F-75724 Paris, France
[3] Univ Gottingen, Max Planck Inst Expt Med, Abt Mol Neurobiol, D-37075 Gottingen, Germany
[4] Univ Gottingen, Max Planck Inst Expt Med, Div Klin Neurowissensch, D-37075 Gottingen, Germany
[5] Deutsches Primatenzentrum, Forschergrp Kognit Ethol, D-37077 Gottingen, Germany
[6] Max Planck Inst Biophys Chem, Biomed NMR Forsch GmbH, D-37077 Gottingen, Germany
[7] Hop Robert Debre, Inst Natl Sante & Rech Med, U676, F-75019 Paris, France
[8] Univ Paris 07, F-75013 Paris, France
关键词
behavior; neuroligin; synaptogenesis;
D O I
10.1073/pnas.0711555105
中图分类号
O [数理科学和化学]; P [天文学、地球科学]; Q [生物科学]; N [自然科学总论];
学科分类号
07 ; 0710 ; 09 ;
摘要
Autism spectrum conditions (ASCs) are heritable conditions characterized by impaired reciprocal social interactions, deficits in language acquisition, and repetitive and restricted behaviors and interests. In addition to more complex genetic susceptibilities, even mutation of a single gene can lead to ASC. Several such monogenic heritable ASC forms are caused by loss-of-function mutations in genes encoding regulators of synapse function in neurons, including NLGN4. We report that mice with a loss-of-function mutation in the murine NLGN4 ortholog Nlgn4, which encodes the synaptic cell adhesion protein Neuroligin-4, exhibit highly selective deficits in reciprocal social interactions and communication that are reminiscent of ASCs in humans. Our findings indicate that a protein network that regulates the maturation and function of synapses in the brain is at the core of a major ASC susceptibility pathway, and establish Neuroligin-4-deficient mice as genetic models for the exploration of the complex neurobiological disorders in ASCs.
引用
收藏
页码:1710 / 1715
页数:6
相关论文
共 43 条
[1]  
BAKKER CE, 1994, CELL, V78, P23
[2]   Fragile X syndrome and autism at the intersection of genetic and neural networks [J].
Belmonte, Matthew K. ;
Bourgeron, Thomas .
NATURE NEUROSCIENCE, 2006, 9 (10) :1221-1225
[3]   Absence of coding mutations in the X-linked genes neuroligin 3 and neuroligin 4 in individuals with autism from the IMGSAC collection [J].
Blasi, F ;
Bacchelli, E ;
Pesaresi, G ;
Carone, S ;
Bailey, AJ ;
Maestrini, E .
AMERICAN JOURNAL OF MEDICAL GENETICS PART B-NEUROPSYCHIATRIC GENETICS, 2006, 141B (03) :220-221
[4]  
Boeckers TM, 1999, J NEUROSCI, V19, P6506
[5]   Subset of individuals with autism spectrum disorders and extreme macrocephaly associated with germline PTEN tumour suppressor gene mutations [J].
Butler, MG ;
Dasouki, MJ ;
Zhou, XP ;
Talebizadeh, Z ;
Brown, M ;
Takahashi, TN ;
Miles, JH ;
Wang, CH ;
Stratton, R ;
Pilarski, R ;
Eng, C .
JOURNAL OF MEDICAL GENETICS, 2005, 42 (04) :318-321
[6]   Fragile X mice develop sensory hyperreactivity to auditory stimuli [J].
Chen, L ;
Toth, M .
NEUROSCIENCE, 2001, 103 (04) :1043-1050
[7]   Control of excitatory and inhibitory synapse formation by neuroligins [J].
Chih, B ;
Engelman, H ;
Scheiffele, P .
SCIENCE, 2005, 307 (5713) :1324-1328
[8]   Activity-dependent validation of excitatory versus inhibitory synapses by neuroligin-1 versus neuroligin-2 [J].
Chubykin, Alexander A. ;
Atasoy, Deniz ;
Etherton, Mark R. ;
Brose, Nils ;
Kavalali, Ege T. ;
Gibson, Jay R. ;
Suedhof, Thomas C. .
NEURON, 2007, 54 (06) :919-931
[9]   Mutations in the gene encoding the synaptic scaffolding protein SHANK3 are associated with autism spectrum disorders [J].
Durand, Christelle M. ;
Betancur, Catalina ;
Boeckers, Tobias M. ;
Bockmann, Juergen ;
Chaste, Pauline ;
Fauchereau, Fabien ;
Nygren, Gudrun ;
Rastam, Maria ;
Gillberg, I. Carina ;
Anckarsater, Henrik ;
Sponheim, Eili ;
Goubran-Botros, Hany ;
Delorme, Richard ;
Chabane, Nadia ;
Mouren-Simeoni, Marie-Christine ;
de Mas, Philippe ;
Bieth, Eric ;
Roge, Bernadette ;
Heron, Delphine ;
Burglen, Lydie ;
Gillberg, Christopher ;
Leboyer, Marion ;
Bourgeron, Thomas .
NATURE GENETICS, 2007, 39 (01) :25-27
[10]  
Fombonne E, 2005, J CLIN PSYCHIAT, V66, P3