The autism- and schizophrenia-associated protein CYFIP1 regulates bilateral brain connectivity and behaviour.

Details

Serval ID
serval:BIB_1AB196467BEA
Type
Article: article from journal or magazin.
Collection
Publications
Institution
Title
The autism- and schizophrenia-associated protein CYFIP1 regulates bilateral brain connectivity and behaviour.
Journal
Nature communications
Author(s)
Domínguez-Iturza N., Lo A.C., Shah D., Armendáriz M., Vannelli A., Mercaldo V., Trusel M., Li K.W., Gastaldo D., Santos A.R., Callaerts-Vegh Z., D'Hooge R., Mameli M., Van der Linden A., Smit A.B., Achsel T., Bagni C.
ISSN
2041-1723 (Electronic)
ISSN-L
2041-1723
Publication state
Published
Issued date
01/08/2019
Peer-reviewed
Oui
Volume
10
Number
1
Pages
3454
Language
english
Notes
Publication types: Journal Article
Publication Status: epublish
Abstract
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.
Pubmed
Web of science
Open Access
Yes
Create date
18/08/2019 15:16
Last modification date
21/08/2019 5:32
Usage data