{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T21:59:02Z","timestamp":1791928742515,"version":"4.3.4"},"reference-count":37,"publisher":"Association for Research in Vision and Ophthalmology (ARVO)","issue":"2","license":[{"start":{"date-parts":[[2020,2,25]],"date-time":"2020-02-25T00:00:00Z","timestamp":1582588800000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Invest. Ophthalmol. Vis. Sci."],"published-print":{"date-parts":[[2020,2,25]]},"DOI":"10.1167\/iovs.61.2.39","type":"journal-article","created":{"date-parts":[[2020,2,25]],"date-time":"2020-02-25T13:31:41Z","timestamp":1582637501000},"page":"39","source":"Crossref","is-referenced-by-count":11,"title":["Modeling\n                    <i>ZNF408<\/i>\n                    -Associated FEVR in Zebrafish Results in Abnormal Retinal Vasculature"],"prefix":"10.1167","volume":"61","author":[{"given":"Dyah W.","family":"Karjosukarso","sequence":"first","affiliation":[{"name":"Department of Human Genetics, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zaheer","family":"Ali","sequence":"additional","affiliation":[{"name":"Unit of Cardiovascular Medicine, Department of Medical and Health Sciences, Link\u00f6ping University, Link\u00f6ping, \u00d6sterg\u00f6tland, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Theo A.","family":"Peters","sequence":"additional","affiliation":[{"name":"Department of Otorhinolaryngology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jia Qi Cheng","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Human Genetics, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anita D M.","family":"Hoogendoorn","sequence":"additional","affiliation":[{"name":"Department of Human Genetics, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alejandro","family":"Garanto","sequence":"additional","affiliation":[{"name":"Department of Human Genetics, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erwin","family":"van Wijk","sequence":"additional","affiliation":[{"name":"Department of Otorhinolaryngology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lasse D.","family":"Jensen","sequence":"additional","affiliation":[{"name":"Unit of Cardiovascular Medicine, Department of Medical and Health Sciences, Link\u00f6ping University, Link\u00f6ping, \u00d6sterg\u00f6tland, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rob W J.","family":"Collin","sequence":"additional","affiliation":[{"name":"Department of Human Genetics, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, Nijmegen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"27","reference":[{"key":"bib1","doi-asserted-by":"publisher","first-page":"247","DOI":"10.1016\/j.preteyeres.2013.07.002","article-title":"Regional morphology and pathophysiology of retinal vascular disease","volume":"36","author":"Bek","year":"2013","journal-title":"Prog Retin Eye Res"},{"key":"bib2","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1038\/eye.2014.70","article-title":"Familial exudative vitreoretinopathy and related retinopathies","volume":"29","author":"Gilmour","year":"2015","journal-title":"Eye (Lond)"},{"key":"bib3","doi-asserted-by":"publisher","first-page":"656","DOI":"10.1002\/humu.v31:6","article-title":"Overview of the mutation spectrum in familial exudative vitreoretinopathy and Norrie disease with identification of 21 novel variants in FZD4, LRP5, and NDP","volume":"31","author":"Nikopoulos","year":"2010","journal-title":"Hum Mutat"},{"key":"bib4","doi-asserted-by":"publisher","first-page":"9856","DOI":"10.1073\/pnas.1220864110","article-title":"ZNF408 is mutated in familial exudative vitreoretinopathy and is crucial for the development of zebrafish retinal vasculature","volume":"110","author":"Collin","year":"2013","journal-title":"Proc Natl Acad Sci U S A"},{"key":"bib5","doi-asserted-by":"publisher","first-page":"1637","DOI":"10.1093\/hmg\/ddw041","article-title":"Haploinsufficiency of RCBTB1 is associated with Coats disease and familial exudative vitreoretinopathy","volume":"25","author":"Wu","year":"2016","journal-title":"Hum Mol Genet"},{"key":"bib6","doi-asserted-by":"publisher","first-page":"468","DOI":"10.3109\/13816810.2015.1120318","article-title":"CTNNB1 mutation associated with familial exudative vitreoretinopathy (FEVR) phenotype","volume":"37","author":"Dixon","year":"2016","journal-title":"Ophthalmic Genet"},{"key":"bib7","doi-asserted-by":"publisher","first-page":"960","DOI":"10.1016\/j.ajhg.2017.05.001","article-title":"Defects in the cell signaling mediator beta-catenin cause the retinal vascular condition FEVR","volume":"100","author":"Panagiotou","year":"2017","journal-title":"Am J Hum Genet"},{"key":"bib8","doi-asserted-by":"publisher","first-page":"68","DOI":"10.1038\/eye.2016.198","article-title":"The zebrafish eye-a paradigm for investigating human ocular genetics","volume":"31","author":"Richardson","year":"2017","journal-title":"Eye (Lond)"},{"key":"bib9","doi-asserted-by":"publisher","first-page":"367","DOI":"10.1038\/eye.2014.19","article-title":"Zebrafish\u2013on the move towards ophthalmological research","volume":"28","author":"Chhetri","year":"2014","journal-title":"Eye (Lond)"},{"key":"bib10","doi-asserted-by":"publisher","first-page":"18","DOI":"10.1186\/s12861-015-0066-9","article-title":"Development and origins of zebrafish ocular vasculature","volume":"15","author":"Kaufman","year":"2015","journal-title":"BMC Dev Biol"},{"key":"bib11","doi-asserted-by":"publisher","first-page":"464","DOI":"10.1016\/j.mod.2009.01.002","article-title":"Small molecule screen for compounds that affect vascular development in the zebrafish retina","volume":"126","author":"Kitambi","year":"2009","journal-title":"Mech Dev"},{"key":"bib12","doi-asserted-by":"publisher","first-page":"114","DOI":"10.1186\/1471-213X-7-114","article-title":"Genetic determinants of hyaloid and retinal vasculature in zebrafish","volume":"7","author":"Alvarez","year":"2007","journal-title":"BMC Dev Biol"},{"key":"bib13","doi-asserted-by":"publisher","first-page":"307","DOI":"10.1006\/dbio.2002.0711","article-title":"In vivo imaging of embryonic vascular development using transgenic zebrafish","volume":"248","author":"Lawson","year":"2002","journal-title":"Dev Biol"},{"key":"bib14","doi-asserted-by":"publisher","first-page":"735","DOI":"10.1016\/j.ydbio.2007.01.023","article-title":"FoxH1 negatively modulates flk1 gene expression and vascular formation in zebrafish","volume":"304","author":"Choi","year":"2007","journal-title":"Dev Biol"},{"key":"bib15","doi-asserted-by":"publisher","first-page":"1735","DOI":"10.1242\/dev.001115","article-title":"Controlling morpholino experiments: don't stop making antisense","volume":"135","author":"Eisen","year":"2008","journal-title":"Development"},{"key":"bib16","doi-asserted-by":"publisher","first-page":"3103","DOI":"10.1242\/dev.112003","article-title":"Out with the old, in with the new: reassessing morpholino knockdowns in light of genome editing technology","volume":"141","author":"Schulte-Merker","year":"2014","journal-title":"Development"},{"key":"bib17","doi-asserted-by":"publisher","first-page":"97","DOI":"10.1016\/j.devcel.2014.11.018","article-title":"Reverse genetic screening reveals poor correlation between morpholino-induced and mutant phenotypes in zebrafish","volume":"32","author":"Kok","year":"2015","journal-title":"Dev Cell"},{"key":"bib18","doi-asserted-by":"publisher","first-page":"e98186","DOI":"10.1371\/journal.pone.0098186","article-title":"Efficient mutagenesis by Cas9 protein-mediated oligonucleotide insertion and large-scale assessment of single-guide RNAs","volume":"9","author":"Gagnon","year":"2014","journal-title":"PLoS One"},{"key":"bib19","doi-asserted-by":"publisher","first-page":"97","DOI":"10.1016\/j.neulet.2007.05.045","article-title":"Expression of the Ku70 subunit (XRCC6) and protection from low dose ionizing radiation during zebrafish embryogenesis","volume":"422","author":"Bladen","year":"2007","journal-title":"Neurosci Lett"},{"key":"bib20","doi-asserted-by":"publisher","first-page":"597","DOI":"10.1089\/zeb.2018.1613","article-title":"Poor Splice-Site Recognition in a Humanized Zebrafish Knockin Model for the Recurrent Deep-Intronic c.7595-2144A&gt;G Mutation in USH2A","volume":"15","author":"Slijkerman","year":"2018","journal-title":"Zebrafish"},{"key":"bib21","doi-asserted-by":"publisher","first-page":"e2748","DOI":"10.1371\/journal.pone.0002748","article-title":"Hypoxia-induced retinal angiogenesis in zebrafish as a model to study retinopathy","volume":"3","author":"Cao","year":"2008","journal-title":"PLoS One"},{"key":"bib22","doi-asserted-by":"publisher","first-page":"1911","DOI":"10.1038\/nprot.2010.150","article-title":"Hypoxia-induced metastasis model in embryonic zebrafish","volume":"5","author":"Rouhi","year":"2010","journal-title":"Nat Protoc"},{"key":"bib23","doi-asserted-by":"publisher","first-page":"3519","DOI":"10.1093\/hmg\/ddy244","article-title":"An FEVR-associated mutation in ZNF408 alters the expression of genes involved in the development of vasculature","volume":"27","author":"Karjosukarso","year":"2018","journal-title":"Hum Mol Genet"},{"key":"bib24","doi-asserted-by":"publisher","first-page":"2220","DOI":"10.1016\/j.ophtha.2014.05.029","article-title":"Diversity of retinal vascular anomalies in patients with familial exudative vitreoretinopathy","volume":"121","author":"Kashani","year":"2014","journal-title":"Ophthalmology"},{"key":"bib25","doi-asserted-by":"publisher","first-page":"50","DOI":"10.1002\/bies.v34.1","article-title":"PRDM proteins: important players in differentiation and disease","volume":"34","author":"Fog","year":"2012","journal-title":"Bioessays"},{"key":"bib26","doi-asserted-by":"publisher","first-page":"2267","DOI":"10.1242\/dev.070110","article-title":"The Prdm family: expanding roles in stem cells and development","volume":"139","author":"Hohenauer","year":"2012","journal-title":"Development"},{"key":"bib27","doi-asserted-by":"publisher","first-page":"883","DOI":"10.1016\/S0092-8674(04)00216-8","article-title":"Vascular development in the retina and inner ear: control by Norrin and Frizzled-4, a high-affinity ligand-receptor pair","volume":"116","author":"Xu","year":"2004","journal-title":"Cell"},{"key":"bib28","doi-asserted-by":"publisher","first-page":"303","DOI":"10.1083\/jcb.200201089","article-title":"Cbfa1-independent decrease in osteoblast proliferation, osteopenia, and persistent embryonic eye vascularization in mice deficient in Lrp5, a Wnt coreceptor","volume":"157","author":"Kato","year":"2002","journal-title":"J Cell Biol"},{"key":"bib29","doi-asserted-by":"publisher","first-page":"299","DOI":"10.1016\/j.cell.2009.07.048","article-title":"TSPAN12 regulates retinal vascular development by promoting Norrin- but not Wnt-induced FZD4\/beta-catenin signaling","volume":"139","author":"Junge","year":"2009","journal-title":"Cell"},{"key":"bib30","doi-asserted-by":"publisher","first-page":"e7867","DOI":"10.1371\/journal.pone.0007867","article-title":"Selective inhibition of retinal angiogenesis by targeting PI3 kinase","volume":"4","author":"Alvarez","year":"2009","journal-title":"PLoS One"},{"key":"bib31","doi-asserted-by":"publisher","first-page":"E5944","DOI":"10.1073\/pnas.1510245112","article-title":"VEGF-B-Neuropilin-1 signaling is spatiotemporally indispensable for vascular and neuronal development in zebrafish","volume":"112","author":"Jensen","year":"2015","journal-title":"Proc Natl Acad Sci U S A"},{"key":"bib32","doi-asserted-by":"publisher","first-page":"2636","DOI":"10.1111\/bph.v174.16","article-title":"Vitamin D receptor agonists regulate ocular developmental angiogenesis and modulate expression of dre-miR-21 and VEGF","volume":"174","author":"Merrigan","year":"2017","journal-title":"Br J Pharmacol"},{"key":"bib33","doi-asserted-by":"publisher","first-page":"494","DOI":"10.1080\/13816810.2016.1275020","article-title":"Exome sequencing confirms ZNF408 mutations as a cause of familial retinitis pigmentosa","volume":"38","author":"Habibi","year":"2017","journal-title":"Ophthalmic Genet"},{"key":"bib34","doi-asserted-by":"publisher","first-page":"4037","DOI":"10.1093\/hmg\/ddv140","article-title":"Whole-exome sequencing reveals ZNF408 as a new gene associated with autosomal recessive retinitis pigmentosa with vitreal alterations","volume":"24","author":"Avila-Fernandez","year":"2015","journal-title":"Hum Mol Genet"},{"key":"bib35","doi-asserted-by":"publisher","first-page":"9675","DOI":"10.1038\/s41598-018-27928-7","article-title":"C2orf71a\/pcare1 is important for photoreceptor outer segment morphogenesis and visual function in zebrafish","volume":"8","author":"Corral-Serrano","year":"2018","journal-title":"Sci Rep"},{"key":"bib36","doi-asserted-by":"publisher","first-page":"148","DOI":"10.1016\/j.exer.2018.05.015","article-title":"Usherin defects lead to early-onset retinal dysfunction in zebrafish","volume":"173","author":"Dona","year":"2018","journal-title":"Exp Eye Res"},{"key":"bib37","doi-asserted-by":"publisher","first-page":"e0200789","DOI":"10.1371\/journal.pone.0200789","article-title":"Eyes shut homolog is important for the maintenance of photoreceptor morphology and visual function in zebrafish","volume":"13","author":"Messchaert","year":"2018","journal-title":"PLoS One"}],"container-title":["Investigative Opthalmology &amp; Visual Science"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/iovs.arvojournals.org\/article.aspx?articleid=2762033","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T21:31:19Z","timestamp":1791927079000},"score":1,"resource":{"primary":{"URL":"https:\/\/iovs.arvojournals.org\/article.aspx?articleid=2762033"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,2,25]]},"references-count":37,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2020,2,7]]}},"URL":"https:\/\/doi.org\/10.1167\/iovs.61.2.39","relation":{},"ISSN":["1552-5783"],"issn-type":[{"value":"1552-5783","type":"print"}],"subject":[],"published":{"date-parts":[[2020,2,25]]}}}