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

Détails

Ressource 1Télécharger: 31371726_BIB_1AB196467BEA.pdf (3976.71 [Ko])
Etat: Public
Version: Final published version
Licence: CC BY 4.0
ID Serval
serval:BIB_1AB196467BEA
Type
Article: article d'un périodique ou d'un magazine.
Collection
Publications
Institution
Titre
The autism- and schizophrenia-associated protein CYFIP1 regulates bilateral brain connectivity and behaviour.
Périodique
Nature communications
Auteur⸱e⸱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
Statut éditorial
Publié
Date de publication
01/08/2019
Peer-reviewed
Oui
Volume
10
Numéro
1
Pages
3454
Langue
anglais
Notes
Publication types: Journal Article
Publication Status: epublish
Résumé
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
Oui
Création de la notice
18/08/2019 15:16
Dernière modification de la notice
30/04/2021 6:08
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