{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T05:31:20Z","timestamp":1790314280917,"version":"4.1.0"},"reference-count":16,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2014,7,3]],"date-time":"2014-07-03T00:00:00Z","timestamp":1404345600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/3.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pigment Cell Melanoma Res"],"published-print":{"date-parts":[[2014,9]]},"abstract":"<jats:title>Summary<\/jats:title><jats:p>Melanin biosynthesis in vertebrates depends on the function of three enzymes of the tyrosinase family, tyrosinase (<jats:styled-content style=\"fixed-case\">T<\/jats:styled-content>yr), tyrosinase\u2010related protein 1 (<jats:styled-content style=\"fixed-case\">T<\/jats:styled-content>yrp1), and dopachrome tautomerase (<jats:styled-content style=\"fixed-case\">D<\/jats:styled-content>ct or <jats:styled-content style=\"fixed-case\">T<\/jats:styled-content>yrp2). <jats:styled-content style=\"fixed-case\">T<\/jats:styled-content>yrp1 might play an additional role in the survival and proliferation of melanocytes. Here, we describe a mutation in <jats:italic>tyrp1<jats:styled-content style=\"fixed-case\">A<\/jats:styled-content><\/jats:italic>, one of the two <jats:italic>tyrp1<\/jats:italic> paralogs in zebrafish, which causes melanophore death leading to a semi\u2010dominant phenotype. The mutation, an <jats:styled-content style=\"fixed-case\">A<\/jats:styled-content>rg\u2010&gt;<jats:styled-content style=\"fixed-case\">C<\/jats:styled-content>ys change in the amino\u2010terminal part of the protein, is similar to mutations in humans and mice where they lead to blond hair (in melanesians) or dark hair with white bases, respectively. We demonstrate that the phenotype in zebrafish depends on the presence of the mutant protein and on melanin synthesis. Ultrastructural analysis shows that the melanosome morphology and pigment content are altered in the mutants. These structural changes might be the underlying cause for the observed cell death, which, surprisingly, does not result in patterning defects.<\/jats:p>","DOI":"10.1111\/pcmr.12272","type":"journal-article","created":{"date-parts":[[2014,6,2]],"date-time":"2014-06-02T10:27:25Z","timestamp":1401704845000},"page":"827-830","source":"Crossref","is-referenced-by-count":45,"title":["A dominant mutation in <i>tyrp1<scp>A<\/scp><\/i> leads to melanophore death in zebrafish"],"prefix":"10.1111","volume":"27","author":[{"given":"Jana","family":"Krauss","sequence":"first","affiliation":[{"name":"Max Planck Institute for Developmental Biology T\u00fcbingen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Silke","family":"Geiger\u2010Rudolph","sequence":"additional","affiliation":[{"name":"Max Planck Institute for Developmental Biology T\u00fcbingen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Iris","family":"Koch","sequence":"additional","affiliation":[{"name":"Max Planck Institute for Developmental Biology T\u00fcbingen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christiane","family":"N\u00fcsslein\u2010Volhard","sequence":"additional","affiliation":[{"name":"Max Planck Institute for Developmental Biology T\u00fcbingen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Uwe","family":"Irion","sequence":"additional","affiliation":[{"name":"Max Planck Institute for Developmental Biology T\u00fcbingen Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2014,7,3]]},"reference":[{"key":"e_1_2_4_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1755-148X.2009.00614.x"},{"key":"e_1_2_4_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0625.2009.00896.x"},{"key":"e_1_2_4_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cub.2014.01.020"},{"key":"e_1_2_4_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/pcmr.12053"},{"key":"e_1_2_4_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/bjc.2013.115"},{"key":"e_1_2_4_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tig.2010.02.002"},{"key":"e_1_2_4_8_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0692-226"},{"key":"e_1_2_4_9_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.123.1.369"},{"key":"e_1_2_4_10_1","doi-asserted-by":"publisher","DOI":"10.1126\/science.1217849"},{"key":"e_1_2_4_11_1","doi-asserted-by":"publisher","DOI":"10.1242\/jcs.017913"},{"key":"e_1_2_4_12_1","doi-asserted-by":"crossref","unstructured":"Oetting W.S.andSetaluri V.(2006).The tyrosinase gene family. In The Pigmentary System: Physiology and Pathophysiology.J.J. Nordlund R.E. Boissy V.J. Hearing R.A. King W.S. Oetting andJ.\u2010P. Ortonneeds.2nd edn. (Oxford:Blackwell Publishing) pp.213\u2013229.","DOI":"10.1002\/9780470987100.ch11"},{"key":"e_1_2_4_13_1","doi-asserted-by":"publisher","DOI":"10.1034\/j.1600-0749.2000.130506.x"},{"key":"e_1_2_4_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/9780470987100.ch14"},{"key":"e_1_2_4_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-1-61779-980-8_14"},{"key":"e_1_2_4_16_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.02533"},{"key":"e_1_2_4_17_1","doi-asserted-by":"publisher","DOI":"10.1093\/genetics\/126.2.443"}],"container-title":["Pigment Cell &amp; Melanoma Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fpcmr.12272","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/pcmr.12272","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/pcmr.12272","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/pcmr.12272","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,24]],"date-time":"2023-09-24T15:55:06Z","timestamp":1695570906000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/pcmr.12272"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,7,3]]},"references-count":16,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2014,9]]}},"alternative-id":["10.1111\/pcmr.12272"],"URL":"https:\/\/doi.org\/10.1111\/pcmr.12272","archive":["Portico"],"relation":{},"ISSN":["1755-1471","1755-148X"],"issn-type":[{"value":"1755-1471","type":"print"},{"value":"1755-148X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2014,7,3]]}}}