New insights into the regulatory function of CYFIP1 in the context of WAVE- and FMRP-containing complexes

dc.contributor.authorAbekhoukh, Sabiha
dc.contributor.authorŞahin, H. Bahar
dc.contributor.authorGrossi, Mauro
dc.contributor.authorZongaro, Samantha
dc.contributor.authorMaurin, Thomas
dc.contributor.authorMadrigal, Irene
dc.contributor.authorKazue-Sugioka, Daniele
dc.contributor.authorRaas-Rothschild, Annick
dc.contributor.authorDoulazmi, Mohamed
dc.contributor.authorCarrera, Pilar
dc.contributor.authorStachon, Andrea
dc.contributor.authorScherer, Steven
dc.contributor.authorDo Nascimento, Maria Rita Drula
dc.contributor.authorTrembleau, Alain
dc.contributor.authorArroyo, Ignacio
dc.contributor.authorSzatmari, Peter
dc.contributor.authorSmith, Isabel M.
dc.contributor.authorMila, Montserrat
dc.contributor.authorSmith, Adam C.
dc.contributor.authorGiangrande, Angela
dc.contributor.authorCaille, Isabelle
dc.contributor.authorBardoni, Barbara
dc.date.accessioned2019-06-27T08:01:22Z
dc.date.available2019-06-27T08:01:22Z
dc.date.issued2017
dc.departmentFakülteler, Mühendislik ve Doğa Bilimleri Fakültesi, Biyoinformatik ve Genetik Bölümüen_US
dc.description.abstractCytoplasmic FMRP interacting protein 1 (CYFIP1) is a candidate gene for intellectual disability (ID) autism schizophrenia and epilepsy. It is a member of a family of proteins that is highly conserved during evolution sharing high homology with its Drosophila homolog dCYFIP. CYFIP1 interacts with the Fragile X mental retardation protein (FMRP encoded by the FMR1 gene) whose absence causes Fragile X syndrome and with the translation initiation factor eIF4E. It is a member of theWAVE regulatory complex (WRC) thus representing a link between translational regulation and the actin cytoskeleton. Here we present data showing a correlation between mRNA levels of CYFIP1 and other members of the WRC. This suggests a tight regulation of the levels of the WRC members not only by post-translational mechanisms as previously hypothesized. Moreover we studied the impact of loss of function of both CYFIP1 and FMRP on neuronal growth and differentiation in two animal models -fly and mouse. We show that these two proteins antagonize each other's function not only during neuromuscular junction growth in the fly but also during new neuronal differentiation in the olfactory bulb of adult mice. Mechanistically FMRP and CYFIP1 modulate mTor signaling in an antagonistic manner likely via independent pathways supporting the results obtained in mouse as well as in fly at the morphological level. Collectively our results illustrate a new model to explain the cellular roles of FMRP and CYFIP1 and the molecular significance of their interaction.en_US]
dc.identifier.citation41
dc.identifier.doi10.1242/dmm.025809en_US
dc.identifier.endpage474
dc.identifier.issn1754-8403en_US
dc.identifier.issn1754-8411en_US
dc.identifier.issn1754-8403
dc.identifier.issn1754-8411
dc.identifier.issue4
dc.identifier.pmid28183735en_US
dc.identifier.scopus2-s2.0-85017508335en_US
dc.identifier.scopusqualityQ1
dc.identifier.startpage463en_US
dc.identifier.urihttps://hdl.handle.net/20.500.12469/356
dc.identifier.urihttps://doi.org/10.1242/dmm.025809
dc.identifier.volume10en_US
dc.identifier.wosWOS:000398899500011en_US
dc.identifier.wosqualityQ1
dc.institutionauthorŞahin, H. Baharen_US
dc.language.isoenen_US
dc.publisherCompany of Biologists Ltden_US
dc.relation.journalDisease Models & Mechanismsen_US
dc.relation.publicationcategoryMakale - Uluslararası Hakemli Dergi - Kurum Öğretim Elemanıen_US
dc.rightsinfo:eu-repo/semantics/openAccessen_US
dc.subjectFragile Xen_US
dc.subjectIntellectual disabilityen_US
dc.subjectAutismen_US
dc.subjectCYFIP1en_US
dc.subjectBP1-BP2 deletionen_US
dc.titleNew insights into the regulatory function of CYFIP1 in the context of WAVE- and FMRP-containing complexesen_US
dc.typeArticleen_US
dspace.entity.typePublication

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