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DOI10.1038/ncomms14042
The autism- and schizophrenia-associated protein CYFIP1 regulates bilateral brain connectivity and behaviour
Dominguez-Iturza, Nuria1; 39;Hooge, Rudi2
2019-08-01
发表期刊NATURE COMMUNICATIONS
ISSN2041-1723
出版年2019
卷号10
文章类型Article
语种英语
国家Switzerland; Belgium; Netherlands; Italy
英文摘要

Copy-number variants of the CYFIP1 gene in humans have been linked to autism spectrum disorders (ASD) and schizophrenia (SCZ), two neuropsychiatric disorders characterized by defects in brain connectivity. Here, we show that CYFIP1 plays an important role in brain functional connectivity and callosal functions. We find that Cyfip1-heterozygous mice have reduced functional connectivity and defects in white matter architecture, similar to phenotypes found in patients with ASD, SCZ and other neuropsychiatric disorders. Cyfip1-deficient mice also present decreased myelination in the callosal axons, altered presynaptic function, and impaired bilateral connectivity. Finally, Cyfip1 deficiency leads to abnormalities in motor coordination, sensorimotor gating and sensory perception, which are also known neuropsychiatric disorder-related symptoms. These results show that Cyfip1 haploinsufficiency compromises brain connectivity and function, which might explain its genetic association to neuropsychiatric disorders.


领域资源环境
收录类别SCI-E ; SSCI
WOS记录号WOS:000478013700002
WOS关键词FRAGILE-X-SYNDROME ; CORPUS-CALLOSUM ; FUNCTIONAL CONNECTIVITY ; SPECTRUM DISORDER ; WHITE-MATTER ; MOUSE MODELS ; PREPULSE INHIBITION ; MOTOR COORDINATION ; AGENESIS ; RISK
WOS类目Multidisciplinary Sciences
WOS研究方向Science & Technology - Other Topics
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文献类型期刊论文
条目标识符http://119.78.100.173/C666/handle/2XK7JSWQ/203165
专题资源环境科学
作者单位1.Univ Lausanne, Dept Fundamental Neurosci, CH-1005 Lausanne, Switzerland;
2.Katholieke Univ Leuven, VIB Ctr Brain & Dis Res, Dept Human Genet, B-3000 Leuven, Belgium;
3.Univ Antwerp, Dept Biomed Sci, Bioimaging Lab, B-2610 Antwerp, Belgium;
4.Katholieke Univ Leuven, Dept Neurosci, VIB Ctr Brain & Dis Res, B-3000 Leuven, Belgium;
5.Katholieke Univ Leuven, Lab Neuro & Psychophysiol, Dept Neurosci, B-3000 Leuven, Belgium;
6.Vrije Univ Amsterdam, Ctr Neurogen & Cognit Res, Dept Mol & Cellular Neurobiol, NL-1081 Amsterdam, Netherlands;
7.Bioincubator, VIB Discovery Sci, B-3001 Heverlee, Belgium;
8.Katholieke Univ Leuven, Lab Biol Psychol, Fac Psychol & Educ Sci, B-3000 Leuven, Belgium;
9.Univ Roma Tor Vergata, Dept Biomed & Prevent, I-00133 Rome, Italy
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Dominguez-Iturza, Nuria,39;Hooge, Rudi. The autism- and schizophrenia-associated protein CYFIP1 regulates bilateral brain connectivity and behaviour[J]. NATURE COMMUNICATIONS,2019,10.
APA Dominguez-Iturza, Nuria,&39;Hooge, Rudi.(2019).The autism- and schizophrenia-associated protein CYFIP1 regulates bilateral brain connectivity and behaviour.NATURE COMMUNICATIONS,10.
MLA Dominguez-Iturza, Nuria,et al."The autism- and schizophrenia-associated protein CYFIP1 regulates bilateral brain connectivity and behaviour".NATURE COMMUNICATIONS 10(2019).
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