{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T05:08:01Z","timestamp":1772168881162,"version":"3.50.1"},"update-to":[{"DOI":"10.12688\/f1000research.15511.1","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2018,12,3]],"date-time":"2018-12-03T00:00:00Z","timestamp":1543795200000}}],"reference-count":21,"publisher":"F1000 Research Ltd","license":[{"start":{"date-parts":[[2018,12,3]],"date-time":"2018-12-03T00:00:00Z","timestamp":1543795200000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000265","name":"Medical Research Council","doi-asserted-by":"publisher","award":["MR\/M012212\/1"],"award-info":[{"award-number":["MR\/M012212\/1"]}],"id":[{"id":"10.13039\/501100000265","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004185","name":"Northern Counties Kidney Research Fund","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004185","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000291","name":"Kidney Research UK","doi-asserted-by":"publisher","award":["PDF_003_20151124"],"award-info":[{"award-number":["PDF_003_20151124"]}],"id":[{"id":"10.13039\/501100000291","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["f1000research.com"],"crossmark-restriction":false},"short-container-title":["F1000Res"],"abstract":"<ns4:p>Zebrafish are a valuable vertebrate model in which to study development and characterize genes involved in cystic kidney disease. Zebrafish embryos and larvae are transparent, allowing non-invasive imaging during their rapid development, which takes place over the first 72 hours post fertilisation. Gene-specific knockdown of nephronophthisis-associated genes leads to ciliary phenotypes which can be assessed in various developmental structures. Here we describe in detail the methods used for imaging cilia within Kupffer\u2019s vesicle to assess nephronophthisis and related ciliopathy phenotypes.<\/ns4:p>","DOI":"10.12688\/f1000research.15511.2","type":"journal-article","created":{"date-parts":[[2018,12,3]],"date-time":"2018-12-03T13:10:09Z","timestamp":1543842609000},"page":"1133","update-policy":"https:\/\/doi.org\/10.12688\/f1000research.crossmark-policy","source":"Crossref","is-referenced-by-count":1,"title":["Using zebrafish to study the function of nephronophthisis and related ciliopathy genes"],"prefix":"10.12688","volume":"7","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1840-915X","authenticated-orcid":false,"given":"Elisa","family":"Molinari","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Simon A.","family":"Ramsbottom","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Veronica","family":"Sammut","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Frances E. P.","family":"Hughes","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1881-3782","authenticated-orcid":false,"given":"John A.","family":"Sayer","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"2560","published-online":{"date-parts":[[2018,12,3]]},"reference":[{"key":"ref-1","doi-asserted-by":"publisher","first-page":"299-304","DOI":"10.1681\/ASN.2004090754","article-title":"Kidney development and disease in the zebrafish.","volume":"16","author":"I Drummond","year":"2005","journal-title":"J Am Soc Nephrol."},{"key":"ref-2","doi-asserted-by":"publisher","first-page":"107-20","DOI":"10.1016\/j.jgg.2016.02.001","article-title":"Zebrafish as a Model for Human Ciliopathies.","volume":"43","author":"Z Song","year":"2016","journal-title":"J Genet Genomics."},{"key":"ref-3","doi-asserted-by":"publisher","first-page":"498-503","DOI":"10.1038\/nature12111","article-title":"The zebrafish reference genome sequence and its relationship to the human genome.","volume":"496","author":"K Howe","year":"2013","journal-title":"Nature."},{"key":"ref-4","doi-asserted-by":"publisher","first-page":"6-15","DOI":"10.4161\/bioa.28014","article-title":"Salient features of the ciliated organ of asymmetry.","volume":"4","author":"J Amack","year":"2014","journal-title":"Bioarchitecture."},{"key":"ref-5","doi-asserted-by":"publisher","first-page":"993-1009","DOI":"10.1007\/s00018-011-0826-z","article-title":"Modelling a ciliopathy: Ahi1 knockdown in model systems reveals an essential role in brain, retinal, and renal development.","volume":"69","author":"R Simms","year":"2012","journal-title":"Cell Mol Life Sci."},{"key":"ref-6","doi-asserted-by":"publisher","first-page":"533-48","DOI":"10.1016\/j.cell.2012.06.028","article-title":"Exome capture reveals ZNF423 and CEP164 mutations, linking renal ciliopathies to DNA damage response signaling.","volume":"150","author":"M Chaki","year":"2012","journal-title":"Cell."},{"key":"ref-7","doi-asserted-by":"publisher","first-page":"3657-66","DOI":"10.1172\/JCI80657","article-title":"DNA replication stress underlies renal phenotypes in CEP290-associated Joubert syndrome.","volume":"125","author":"G Slaats","year":"2015","journal-title":"J Clin Invest."},{"key":"ref-8","doi-asserted-by":"publisher","first-page":"406-16","DOI":"10.1038\/ejhg.2008.238","article-title":"Nephronophthisis.","volume":"17","author":"R Simms","year":"2009","journal-title":"Eur J Hum Genet."},{"key":"ref-9","doi-asserted-by":"publisher","first-page":"181-94","DOI":"10.1007\/s00467-010-1585-z","article-title":"Nephronophthisis.","volume":"26","author":"M Wolf","year":"2011","journal-title":"Pediatr Nephrol."},{"key":"ref-10","doi-asserted-by":"publisher","first-page":"287","DOI":"10.3389\/fped.2017.00287","article-title":"Many Genes-One Disease? Genetics of Nephronophthisis (NPHP) and NPHP-Associated Disorders.","volume":"5","author":"S Srivastava","year":"2018","journal-title":"Front Pediatr."},{"key":"ref-11","doi-asserted-by":"publisher","first-page":"928-40","DOI":"10.1038\/nrg1727","article-title":"Cilia and centrosomes: a unifying pathogenic concept for cystic kidney disease?","volume":"6","author":"F Hildebrandt","year":"2005","journal-title":"Nat Rev Genet."},{"key":"ref-12","doi-asserted-by":"publisher","first-page":"317-9","DOI":"10.1016\/j.tcb.2015.03.005","article-title":"Are renal ciliopathies (replication) stressed out?","volume":"25","author":"G Slaats","year":"2015","journal-title":"Trends Cell Biol."},{"key":"ref-13","doi-asserted-by":"publisher","first-page":"37-8","DOI":"10.1038\/418037a","article-title":"Conserved function for embryonic nodal cilia.","volume":"418","author":"J Essner","year":"2002","journal-title":"Nature."},{"key":"ref-14","doi-asserted-by":"publisher","first-page":"1907-21","DOI":"10.1242\/dev.01772","article-title":"Cilia-driven fluid flow in the zebrafish pronephros, brain and Kupffer's vesicle is required for normal organogenesis.","volume":"132","author":"A Kramer-Zucker","year":"2005","journal-title":"Development."},{"key":"ref-15","doi-asserted-by":"publisher","DOI":"10.3791\/50038","article-title":"Analysis of gene function and visualization of cilia-generated fluid flow in Kupffer's vesicle.","author":"G Wang","year":"2013","journal-title":"J Vis Exp."},{"key":"ref-16","doi-asserted-by":"publisher","first-page":"705-25","DOI":"10.1007\/s00285-016-0967-7","article-title":"Three-dimensional flow in Kupffer's Vesicle.","volume":"73","author":"T Montenegro-Johnson","year":"2016","journal-title":"J Math Biol."},{"key":"ref-17","doi-asserted-by":"publisher","first-page":"119-25","DOI":"10.4161\/19490992.2014.956593","article-title":"Organized chaos in Kupffer's vesicle: how a heterogeneous structure achieves consistent left-right patterning.","volume":"4","author":"D Smith","year":"2014","journal-title":"Bioarchitecture."},{"key":"ref-18","doi-asserted-by":"publisher","first-page":"45","DOI":"10.1186\/s12929-017-0351-y","article-title":"Klf8 regulates left-right asymmetric patterning through modulation of Kupffer's vesicle morphogenesis and spaw expression.","volume":"24","author":"C Lin","year":"2017","journal-title":"J Biomed Sci."},{"key":"ref-19","doi-asserted-by":"publisher","first-page":"e53162","DOI":"10.3791\/53162","article-title":"Using Confocal Analysis of Xenopus laevis to Investigate Modulators of Wnt and Shh Morphogen Gradients.","author":"S Fellgett","year":"2015","journal-title":"J Vis Exp."},{"key":"ref-20","doi-asserted-by":"publisher","first-page":"841-51","DOI":"10.1038\/ncb3029","article-title":"Prostaglandin signalling regulates ciliogenesis by modulating intraflagellar transport.","volume":"16","author":"D Jin","year":"2014","journal-title":"Nat Cell Biol."},{"key":"ref-21","article-title":"Dataset 1 in: Using zebrafish to study the function of nephronophthisis and related ciliopathy genes.","author":"E Molinari","year":"2018","journal-title":"F1000Research."}],"container-title":["F1000Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/f1000research.com\/articles\/7-1133\/v2\/pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/f1000research.com\/articles\/7-1133\/v2\/xml","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/f1000research.com\/articles\/7-1133\/v2\/iparadigms","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2018,12,6]],"date-time":"2018-12-06T14:21:13Z","timestamp":1544106073000},"score":1,"resource":{"primary":{"URL":"https:\/\/f1000research.com\/articles\/7-1133\/v2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,12,3]]},"references-count":21,"URL":"https:\/\/doi.org\/10.12688\/f1000research.15511.2","relation":{"has-review":[{"id-type":"doi","id":"10.5256\/f1000research.18693.r41419","asserted-by":"object"},{"id-type":"doi","id":"10.5256\/f1000research.18693.r41420","asserted-by":"object"}]},"ISSN":["2046-1402"],"issn-type":[{"value":"2046-1402","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,12,3]]},"assertion":[{"value":"Indexed","URL":"https:\/\/f1000research.com\/articles\/7-1133\/v2#article-reports","order":0,"name":"referee-status","label":"Referee status","group":{"name":"current-referee-status","label":"Current Referee Status"}},{"value":"10.5256\/f1000research.16913.r36493, Weibin Zhou, Duke University, Durham, NC, USA, 13 Aug 2018, version 1, indexed","URL":"https:\/\/f1000research.com\/articles\/7-1133\/v1#referee-response-36493","order":0,"name":"referee-response-36493","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"10.5256\/f1000research.16913.r37381, Glenn P. Lobo, School of Medicine, Department of Ophthalmology, Medical University of South Carolina (MUSC), Charleston, SC, USA, 30 Aug 2018, version 1, indexed","URL":"https:\/\/f1000research.com\/articles\/7-1133\/v1#referee-response-37381","order":1,"name":"referee-response-37381","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"10.5256\/f1000research.16913.r37382, Roslyn Simms, University of Sheffield, Sheffield, UK, 05 Sep 2018, version 1, indexed","URL":"https:\/\/f1000research.com\/articles\/7-1133\/v1#referee-response-37382","order":2,"name":"referee-response-37382","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"This work was supported by Northern Counties Kidney Research Fund, Kidney Research UK (PDF_003_20151124) and the Medical Research Council (MR\/M012212\/1). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.","order":3,"name":"grant-information","label":"Grant Information"},{"value":"This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.Data associated with the article are available under the terms of the Creative Commons Zero \"No rights reserved\" data waiver (CC0 1.0 Public domain dedication).","order":0,"name":"copyright-info","label":"Copyright"}]}}