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Mutations in\n                    <jats:italic>RBM10<\/jats:italic>\n                    are known to cause multiple congenital anomaly syndrome in male humans, the TARP syndrome. However, the molecular function of RBM10 is unknown. Here we used PAR\u2010CLIP to identify thousands of binding sites of RBM10 and observed significant RBM10\u2013RNA interactions in the vicinity of splice sites. Computational analyses of binding sites as well as loss\u2010of\u2010function and gain\u2010of\u2010function experiments provided evidence for the function of RBM10 in regulating exon skipping and suggested an underlying mechanistic model, which could be subsequently validated by minigene experiments. Furthermore, we demonstrated the splicing defects in a patient carrying an\n                    <jats:italic>RBM10<\/jats:italic>\n                    mutation, which could be explained by disrupted function of RBM10 in splicing regulation. Overall, our study established RBM10 as an important regulator of alternative splicing, presented a mechanistic model for RBM10\u2010mediated splicing regulation and provided a molecular link to understanding a human congenital disorder.\n                  <\/jats:p>","DOI":"10.1002\/emmm.201302663","type":"journal-article","created":{"date-parts":[[2013,8,22]],"date-time":"2013-08-22T07:24:53Z","timestamp":1377156293000},"page":"1431-1442","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":104,"title":["Integrative analysis revealed the molecular mechanism underlying RBM10\u2010mediated splicing regulation"],"prefix":"10.1038","volume":"5","author":[{"given":"Yongbo","family":"Wang","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andreas","family":"Gogol\u2010D\u00f6ring","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hao","family":"Hu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sebastian","family":"Fr\u00f6hler","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yunxia","family":"Ma","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marvin","family":"Jens","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jonas","family":"Maaskola","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasuhiro","family":"Murakawa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claudia","family":"Quedenau","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Markus","family":"Landthaler","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vera","family":"Kalscheuer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dagmar","family":"Wieczorek","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yang","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuhui","family":"Hu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wei","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2013,8,22]]},"reference":[{"key":"BFEMMM201302663_CR1","doi-asserted-by":"crossref","first-page":"2667","DOI":"10.1128\/MCB.05266-11","volume":"31","author":"DE Agafonov","year":"2011","unstructured":"Agafonov DE, Deckert J, Wolf E, Odenw\u00e4lder P, Bessonov S, Will CL, Urlaub H, L\u00fchrmann R (2011) Semiquantitative proteomic analysis of the human spliceosome via a novel two\u2010dimensional gel electrophoresis method. 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