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A subset of patients exhibit social behavioural problems that meet the diagnostic criteria for autism spectrum disorder (ASD); however, the underlying molecular pathogenesis remains poorly understood. <jats:italic>PX-RICS<\/jats:italic> is located in the chromosomal region commonly deleted in JBS patients with autistic-like behaviour. Here we report that <jats:italic>PX-RICS<\/jats:italic>-deficient mice exhibit ASD-like social behaviours and ASD-related comorbidities. <jats:italic>PX-RICS<\/jats:italic>-deficient neurons show reduced surface \u03b3-aminobutyric acid type A receptor (GABA<jats:sub>A<\/jats:sub>R) levels and impaired GABA<jats:sub>A<\/jats:sub>R-mediated synaptic transmission. PX-RICS, GABARAP and 14-3-3\u03b6\/\u03b8 form an adaptor complex that interconnects GABA<jats:sub>A<\/jats:sub>R and dynein\/dynactin, thereby facilitating GABA<jats:sub>A<\/jats:sub>R surface expression. ASD-like behavioural abnormalities in <jats:italic>PX-RICS<\/jats:italic>-deficient mice are ameliorated by enhancing inhibitory synaptic transmission with a GABA<jats:sub>A<\/jats:sub>R agonist. Our findings demonstrate a critical role of PX-RICS in cognition and suggest a causal link between <jats:italic>PX-RICS<\/jats:italic> deletion and ASD-like behaviour in JBS patients.<\/jats:p>","DOI":"10.1038\/ncomms10861","type":"journal-article","created":{"date-parts":[[2016,3,16]],"date-time":"2016-03-16T11:46:48Z","timestamp":1458128808000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":57,"title":["PX-RICS-deficient mice mimic autism spectrum disorder in Jacobsen syndrome through impaired GABAA receptor trafficking"],"prefix":"10.1038","volume":"7","author":[{"given":"Tsutomu","family":"Nakamura","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fumiko","family":"Arima-Yoshida","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fumika","family":"Sakaue","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yukiko","family":"Nasu-Nishimura","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasuko","family":"Takeda","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ken","family":"Matsuura","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natacha","family":"Akshoomoff","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah N.","family":"Mattson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul D.","family":"Grossfeld","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Toshiya","family":"Manabe","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tetsu","family":"Akiyama","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2016,3,16]]},"reference":[{"key":"BFncomms10861_CR1","doi-asserted-by":"publisher","first-page":"24","DOI":"10.1016\/j.cell.2011.09.017","volume":"147","author":"C Lord","year":"2011","unstructured":"Lord, C. 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