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The cytoplasmic FMRP-interacting protein 1 (<jats:italic>CYFIP1<\/jats:italic>) is one of the four genes contained within this locus and has been associated with other genetic forms of autism spectrum disorders (ASD). In mice, <jats:italic>Cyfip1<\/jats:italic> haploinsufficiency leads to alteration of dendritic spine morphology and defects in synaptic plasticity, two pathophysiological hallmarks of mouse models of ASD. At the behavioral level, however, <jats:italic>Cyfip1<\/jats:italic> haploinsufficiency leads to minor phenotypes, not directly relevant for 15q11.2 deletion syndrome or ASD. A fundamental question is whether neuronal phenotypes caused by the mutation of <jats:italic>Cyfip1<\/jats:italic> are relevant for the human condition. Here, we describe a synaptic cluster of ASD-associated proteins centered on CYFIP1 and the adhesion protein Neuroligin-3. <jats:italic>Cyfip1<\/jats:italic> haploinsufficiency in mice led to decreased dendritic spine density and stability associated with social behavior and motor learning phenotypes. Behavioral training early in development resulted in alleviating the motor learning deficits caused by <jats:italic>Cyfip1<\/jats:italic> haploinsufficiency. Altogether, these data provide new insight into the neuronal and behavioral phenotypes caused by <jats:italic>Cyfip1<\/jats:italic> mutation and proof-of-concept for the development of a behavioral therapy to treat phenotypes associated with 15q11.2 syndromes and ASD.<\/jats:p>","DOI":"10.1038\/s41398-018-0338-9","type":"journal-article","created":{"date-parts":[[2019,1,21]],"date-time":"2019-01-21T12:03:16Z","timestamp":1548072196000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":26,"title":["Behavioral training rescues motor deficits in Cyfip1 haploinsufficiency mouse model of autism spectrum disorders"],"prefix":"10.1038","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7917-5799","authenticated-orcid":false,"given":"Sven O.","family":"Bachmann","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Monika","family":"Sledziowska","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ellen","family":"Cross","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shireene","family":"Kalbassi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sophie","family":"Waldron","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fangli","family":"Chen","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4804-0832","authenticated-orcid":false,"given":"Adam","family":"Ranson","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6902-6071","authenticated-orcid":false,"given":"St\u00e9phane J.","family":"Baudouin","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2019,1,21]]},"reference":[{"key":"338_CR1","doi-asserted-by":"publisher","first-page":"896","DOI":"10.1016\/S0140-6736(13)61539-1","volume":"383","author":"MC Lai","year":"2014","unstructured":"Lai, M. 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