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Recently, paternally derived duplication has also been shown to contribute to the development of ASD. The molecular mechanism underlying paternal Dup15q remains unclear. Here, we conduct genetic and overexpression-based screening and identify\n                    <jats:italic>Necdin<\/jats:italic>\n                    (\n                    <jats:italic>Ndn<\/jats:italic>\n                    ) as a driver gene for paternal Dup15q resulting in the development of ASD-like phenotypes in mice. An excess amount of\n                    <jats:italic>Ndn<\/jats:italic>\n                    results in enhanced spine formation and density as well as hyperexcitability of cortical pyramidal neurons. We generate\n                    <jats:italic>15q dup\u0394Ndn<\/jats:italic>\n                    mice with a normalized copy number of\n                    <jats:italic>Ndn<\/jats:italic>\n                    by excising its one copy from Dup15q mice using a CRISPR-Cas9 system.\n                    <jats:italic>15q dup\u0394Ndn<\/jats:italic>\n                    mice do not show ASD-like phenotypes and show dendritic spine dynamics and cortical excitatory-inhibitory balance similar to wild type animals. Our study provides an insight into the role of\n                    <jats:italic>Ndn<\/jats:italic>\n                    in paternal 15q duplication and a mouse model of paternal Dup15q syndrome.\n                  <\/jats:p>","DOI":"10.1038\/s41467-021-24359-3","type":"journal-article","created":{"date-parts":[[2021,7,1]],"date-time":"2021-07-01T06:05:24Z","timestamp":1625119524000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":29,"title":["Genetic dissection identifies Necdin as a driver gene in a mouse model of paternal 15q duplications"],"prefix":"10.1038","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6575-6189","authenticated-orcid":false,"given":"Kota","family":"Tamada","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5626-9445","authenticated-orcid":false,"given":"Keita","family":"Fukumoto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tsuyoshi","family":"Toya","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7171-230X","authenticated-orcid":false,"given":"Nobuhiro","family":"Nakai","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janak R.","family":"Awasthi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shinji","family":"Tanaka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1216-8890","authenticated-orcid":false,"given":"Shigeo","family":"Okabe","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1960-829X","authenticated-orcid":false,"given":"Fran\u00e7ois","family":"Spitz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5625-8460","authenticated-orcid":false,"given":"Fumihito","family":"Saitow","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7133-4302","authenticated-orcid":false,"given":"Hidenori","family":"Suzuki","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7153-266X","authenticated-orcid":false,"given":"Toru","family":"Takumi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,7,1]]},"reference":[{"key":"24359_CR1","doi-asserted-by":"publisher","first-page":"1","DOI":"10.15585\/mmwr.ss6706a1","volume":"67","author":"J Baio","year":"2018","unstructured":"Baio, J. et al. 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