IDEAS home Printed from https://ideas.repec.org/a/nat/natcom/v10y2019i1d10.1038_s41467-019-11203-y.html
   My bibliography  Save this article

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

Author

Listed:
  • Nuria Domínguez-Iturza

    (University of Lausanne
    VIB Center for Brain & Disease Research)

  • Adrian C. Lo

    (University of Lausanne)

  • Disha Shah

    (University of Antwerp
    VIB Center for Brain & Disease Research)

  • Marcelo Armendáriz

    (KU Leuven)

  • Anna Vannelli

    (University of Lausanne)

  • Valentina Mercaldo

    (University of Lausanne)

  • Massimo Trusel

    (University of Lausanne)

  • Ka Wan Li

    (VU University Amsterdam)

  • Denise Gastaldo

    (University of Lausanne)

  • Ana Rita Santos

    (VIB Center for Brain & Disease Research
    VIB Discovery Sciences, Bioincubator)

  • Zsuzsanna Callaerts-Vegh

    (Laboratory of Biological Psychology)

  • Rudi D’Hooge

    (Laboratory of Biological Psychology)

  • Manuel Mameli

    (University of Lausanne)

  • Annemie Van der Linden

    (University of Antwerp)

  • August B. Smit

    (VU University Amsterdam)

  • Tilmann Achsel

    (University of Lausanne
    VIB Center for Brain & Disease Research)

  • Claudia Bagni

    (University of Lausanne
    VIB Center for Brain & Disease Research
    University of Rome Tor Vergata)

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.

Suggested Citation

  • Nuria Domínguez-Iturza & Adrian C. Lo & Disha Shah & Marcelo Armendáriz & Anna Vannelli & Valentina Mercaldo & Massimo Trusel & Ka Wan Li & Denise Gastaldo & Ana Rita Santos & Zsuzsanna Callaerts-Vegh, 2019. "The autism- and schizophrenia-associated protein CYFIP1 regulates bilateral brain connectivity and behaviour," Nature Communications, Nature, vol. 10(1), pages 1-13, December.
    • Handle: RePEc:nat:natcom:v:10:y:2019:i:1:d:10.1038_s41467-019-11203-y
    DOI: 10.1038/s41467-019-11203-y
    as

    Download full text from publisher

    File URL: https://www.nature.com/articles/s41467-019-11203-y
    File Function: Abstract
    Download Restriction: no
    File URL: https://libkey.io/10.1038/s41467-019-11203-y?utm_source=ideas
    LibKey link: if access is restricted and if your library uses this service, LibKey will redirect you to where you can use your library subscription to access this item
    ---><---

    Citations

    Citations are extracted by the CitEc Project, subscribe to its RSS feed for this item.
    as


    Cited by:

    1. Vittoria Mariano & Alexandros K. Kanellopoulos & Giuseppe Aiello & Adrian C. Lo & Eric Legius & Tilmann Achsel & Claudia Bagni, 2023. "SREBP modulates the NADP+/NADPH cycle to control night sleep in Drosophila," Nature Communications, Nature, vol. 14(1), pages 1-15, December.

    More about this item

    Statistics

    Access and download statistics

    Corrections

    All material on this site has been provided by the respective publishers and authors. You can help correct errors and omissions. When requesting a correction, please mention this item's handle: RePEc:nat:natcom:v:10:y:2019:i:1:d:10.1038_s41467-019-11203-y. See general information about how to correct material in RePEc.

    If you have authored this item and are not yet registered with RePEc, we encourage you to do it here. This allows to link your profile to this item. It also allows you to accept potential citations to this item that we are uncertain about.

    We have no bibliographic references for this item. You can help adding them by using this form .

    If you know of missing items citing this one, you can help us creating those links by adding the relevant references in the same way as above, for each refering item. If you are a registered author of this item, you may also want to check the "citations" tab in your RePEc Author Service profile, as there may be some citations waiting for confirmation.

    For technical questions regarding this item, or to correct its authors, title, abstract, bibliographic or download information, contact: Sonal Shukla or Springer Nature Abstracting and Indexing (email available below). General contact details of provider: http://www.nature.com .

    Please note that corrections may take a couple of weeks to filter through the various RePEc services.

    IDEAS is a RePEc service. RePEc uses bibliographic data supplied by the respective publishers.