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Here, we present zebrafish models of <jats:styled-content style=\"fixed-case\">UM<\/jats:styled-content>, which are driven by melanocyte\u2010specific expression of activating <jats:italic><jats:styled-content style=\"fixed-case\">GNAQ<\/jats:styled-content><\/jats:italic> or <jats:italic><jats:styled-content style=\"fixed-case\">GNA<\/jats:styled-content>11<\/jats:italic> alleles<jats:italic>, <jats:styled-content style=\"fixed-case\">GNAQ<\/jats:styled-content>\/11<\/jats:italic><jats:sup><jats:italic>Q209L<\/jats:italic><\/jats:sup>, the predominant initiating mutations for human <jats:styled-content style=\"fixed-case\">UM<\/jats:styled-content>. When combined with mutant <jats:italic>tp53<\/jats:italic>,<jats:italic> <jats:styled-content style=\"fixed-case\">GNAQ<\/jats:styled-content>\/11<\/jats:italic><jats:sup><jats:italic>Q209L<\/jats:italic><\/jats:sup> transgenics develop various melanocytic tumors, including <jats:styled-content style=\"fixed-case\">UM<\/jats:styled-content>, with near complete penetrance. These tumors display nuclear <jats:styled-content style=\"fixed-case\">YAP<\/jats:styled-content> localization and thus phenocopy human <jats:styled-content style=\"fixed-case\">UM<\/jats:styled-content>. We show that <jats:italic><jats:styled-content style=\"fixed-case\">GNAQ<\/jats:styled-content>\/11<\/jats:italic><jats:sup><jats:italic>Q209L<\/jats:italic><\/jats:sup> expression induces profound melanocyte defects independent of <jats:italic>tp53<\/jats:italic> mutation, which are apparent within 3\u00a0days of development. First, increases in melanocyte number, melanin content, and subcellular melanin distribution result in hyperpigmentation. Additionally, altered melanocyte migration, survival properties, and evasion of normal boundary cues lead to aberrant melanocyte localization and stripe patterning. Collectively, these data show that <jats:italic><jats:styled-content style=\"fixed-case\">GNAQ<\/jats:styled-content>\/11<\/jats:italic><jats:sup><jats:italic>Q209L<\/jats:italic><\/jats:sup> is sufficient to induce numerous protumorigenic changes within melanocytes.<\/jats:p>","DOI":"10.1111\/pcmr.12700","type":"journal-article","created":{"date-parts":[[2018,3,23]],"date-time":"2018-03-23T19:05:52Z","timestamp":1521831952000},"page":"604-613","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":28,"title":["Uveal melanoma driver mutations in <scp>GNAQ<\/scp>\/11 yield numerous changes in melanocyte biology"],"prefix":"10.1111","volume":"31","author":[{"given":"Dahlia E.","family":"Perez","sequence":"first","affiliation":[{"name":"Department of Biology and The David H. Koch Institute for Integrative Cancer Research Massachusetts Institute of Technology  Cambridge MA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andrea M.","family":"Henle","sequence":"additional","affiliation":[{"name":"Department of Biology and The David H. Koch Institute for Integrative Cancer Research Massachusetts Institute of Technology  Cambridge MA USA"},{"name":"Biology Department Carthage College  Kenosha WI USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Adam","family":"Amsterdam","sequence":"additional","affiliation":[{"name":"Department of Biology and The David H. Koch Institute for Integrative Cancer Research Massachusetts Institute of Technology  Cambridge MA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hannah R.","family":"Hagen","sequence":"additional","affiliation":[{"name":"Department of Biology and The David H. Koch Institute for Integrative Cancer Research Massachusetts Institute of Technology  Cambridge MA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jacqueline A.","family":"Lees","sequence":"additional","affiliation":[{"name":"Department of Biology and The David H. Koch Institute for Integrative Cancer Research Massachusetts Institute of Technology  Cambridge MA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2018,4,6]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.13.20.2713"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0406252102"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.019299"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1167\/iovs.14-14550"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ccr.2014.04.016"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.1777309"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0067801"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1158\/0008-5472.CAN-14-3229"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/pcmr.12482"},{"key":"e_1_2_7_11_1","first-page":"1230","article-title":"Ocular melanoma: An overview of the current status","volume":"6","author":"Jovanovic P.","year":"2013","journal-title":"International Journal of Clinical and Experimental Pathology"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.semcdb.2008.10.001"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1080280"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0749.2006.00307.x"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.004747"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.106567"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.18632\/oncotarget.9207"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1167\/iovs.08-2145"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/pcmr.12332"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2005.01.031"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10544-014-9908-7"},{"key":"e_1_2_7_22_1","first-page":"5712","article-title":"Lack of BRAF Mutations in Uveal Melanoma","volume":"63","author":"Rimoldi D.","year":"2003","journal-title":"Cancer Research"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1097\/CMR.0000000000000121"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-319-22539-5_12"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2014.11.013"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature07586"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1000584"},{"key":"e_1_2_7_28_1","doi-asserted-by":"publisher","DOI":"10.1038\/icb.2014.53"},{"key":"e_1_2_7_29_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrc3589"},{"key":"e_1_2_7_30_1","doi-asserted-by":"publisher","DOI":"10.1158\/1535-7163.MCT-12-0121"},{"key":"e_1_2_7_31_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1315416111"},{"key":"e_1_2_7_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ccell.2016.04.015"},{"key":"e_1_2_7_33_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ccr.2014.04.017"}],"container-title":["Pigment Cell &amp; 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