{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T18:24:19Z","timestamp":1790619859309,"version":"4.1.0"},"reference-count":46,"publisher":"Elsevier BV","issue":"3","license":[{"start":{"date-parts":[[2024,3,1]],"date-time":"2024-03-01T00:00:00Z","timestamp":1709251200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2024,3,1]],"date-time":"2024-03-01T00:00:00Z","timestamp":1709251200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2024,2,8]],"date-time":"2024-02-08T00:00:00Z","timestamp":1707350400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["EY034497"],"award-info":[{"award-number":["EY034497"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["EY014800"],"award-info":[{"award-number":["EY014800"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001116","name":"Foundation Fighting Blindness Inc","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001116","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007747","name":"University of Utah","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007747","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000262","name":"Foundation Fighting Blindness","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000262","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001818","name":"Research to Prevent Blindness","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001818","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["jlr.org","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Journal of Lipid Research"],"published-print":{"date-parts":[[2024,3]]},"DOI":"10.1016\/j.jlr.2024.100518","type":"journal-article","created":{"date-parts":[[2024,2,9]],"date-time":"2024-02-09T21:45:42Z","timestamp":1707515142000},"page":"100518","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":8,"title":["Elovl4b knockout\u00a0zebrafish as a model for ocular very-long-chain PUFA deficiency"],"prefix":"10.1016","volume":"65","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7122-7307","authenticated-orcid":false,"given":"Uzoamaka","family":"Nwagbo","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8608-1494","authenticated-orcid":false,"given":"Saba","family":"Parvez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3217-2299","authenticated-orcid":false,"given":"J. Alan","family":"Maschek","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4228-7666","authenticated-orcid":false,"given":"Paul S.","family":"Bernstein","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.jlr.2024.100518_bib1","doi-asserted-by":"crossref","first-page":"3096","DOI":"10.3390\/nu15143096","article-title":"Understanding the roles of very-long-chain polyunsaturated fatty acids (VLC-PUFAs) in eye health","volume":"15","author":"Nwagbo","year":"2023","journal-title":"Nutrients"},{"key":"10.1016\/j.jlr.2024.100518_bib2","doi-asserted-by":"crossref","first-page":"3217","DOI":"10.1194\/jlr.M007518","article-title":"Long-chain and very long-chain polyunsaturated fatty acids in ocular aging and age-related macular degeneration","volume":"51","author":"Liu","year":"2010","journal-title":"J.\u00a0Lipid Res."},{"key":"10.1016\/j.jlr.2024.100518_bib3","doi-asserted-by":"crossref","first-page":"5563","DOI":"10.1039\/D1OB00491C","article-title":"The synthesis of the very long chain polyunsaturated fatty acid (VLC-PUFA) 32:6 n-3","volume":"19","author":"Wade","year":"2021","journal-title":"Org. Biomol. Chem."},{"key":"10.1016\/j.jlr.2024.100518_bib4","doi-asserted-by":"crossref","DOI":"10.1002\/mnfr.201801058","article-title":"n-3 PUFA supplementation alters retinal very-long-chain-PUFA levels and ratios in diabetic animal models","volume":"63","author":"Gorusupudi","year":"2019","journal-title":"Mol. Nutr. Food Res."},{"key":"10.1016\/j.jlr.2024.100518_bib5","doi-asserted-by":"crossref","first-page":"3669","DOI":"10.1167\/iovs.13-13099","article-title":"Mutant ELOVL4 that causes autosomal dominant stargardt-3 macular dystrophy is misrouted to rod outer segment disks","volume":"55","author":"Agbaga","year":"2014","journal-title":"Invest. Ophthalmol. Vis. Sci."},{"key":"10.1016\/j.jlr.2024.100518_bib6","doi-asserted-by":"crossref","first-page":"4273","DOI":"10.1073\/pnas.0608142104","article-title":"A\u00a0female-biased expressed elongase involved in long-chain hydrocarbon biosynthesis and courtship behavior in Drosophila melanogaster","volume":"104","author":"Chertemps","year":"2007","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.jlr.2024.100518_bib7","doi-asserted-by":"crossref","DOI":"10.1111\/acel.13100","article-title":"The lipid elongation enzyme ELOVL2 is a molecular regulator of aging in the retina","volume":"19","author":"Chen","year":"2020","journal-title":"Aging Cell"},{"key":"10.1016\/j.jlr.2024.100518_bib8","doi-asserted-by":"crossref","first-page":"745","DOI":"10.1016\/j.ajhg.2011.10.011","article-title":"Recessive mutations in ELOVL4 cause ichthyosis, intellectual disability, and spastic quadriplegia","volume":"89","author":"Aldahmesh","year":"2011","journal-title":"Am. J. Hum. Genet."},{"key":"10.1016\/j.jlr.2024.100518_bib9","doi-asserted-by":"crossref","first-page":"1795","DOI":"10.1007\/s12035-017-0824-8","article-title":"Homozygous expression of mutant ELOVL4 leads to seizures and death in a novel animal model of very long-chain fatty acid deficiency","volume":"55","author":"Hopiavuori","year":"2018","journal-title":"Mol. Neurobiol."},{"key":"10.1016\/j.jlr.2024.100518_bib10","doi-asserted-by":"crossref","first-page":"1145","DOI":"10.1016\/j.bbalip.2010.06.005","article-title":"Expression and role of Elovl4 elongases in biosynthesis of very long-chain fatty acids during zebrafish Danio rerio early embryonic development","volume":"1801","author":"Monroig","year":"2010","journal-title":"Biochim. Biophys. Acta"},{"key":"10.1016\/j.jlr.2024.100518_bib11","doi-asserted-by":"crossref","first-page":"120","DOI":"10.7150\/ijbs.3.120","article-title":"Depletion of ceramides with very long chain fatty acids causes defective skin permeability barrier function, and neonatal lethality in ELOVL4 deficient mice","volume":"3","author":"Li","year":"2007","journal-title":"Int. J. Biol. Sci."},{"key":"10.1016\/j.jlr.2024.100518_bib12","doi-asserted-by":"crossref","first-page":"4164","DOI":"10.1073\/pnas.0407698102","article-title":"Lipofuscin accumulation, abnormal electrophysiology, and photoreceptor degeneration in mutant ELOVL4 transgenic mice: a model for macular degeneration","volume":"102","author":"Karan","year":"2005","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.jlr.2024.100518_bib13","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1016\/j.preteyeres.2018.10.004","article-title":"ELOVL4: very long-chain fatty acids serve an eclectic role in mammalian health and function","volume":"69","author":"Hopiavuori","year":"2019","journal-title":"Prog. Retin. Eye Res."},{"key":"10.1016\/j.jlr.2024.100518_bib14","doi-asserted-by":"crossref","DOI":"10.1073\/pnas.2017739118","article-title":"Retinal bioavailability and functional effects of a synthetic very-long-chain polyunsaturated fatty acid in mice","volume":"118","author":"Gorusupudi","year":"2021","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.jlr.2024.100518_bib15","doi-asserted-by":"crossref","first-page":"122","DOI":"10.1016\/j.cbpb.2011.02.007","article-title":"Biosynthesis of very long-chain fatty acids (C > 24) in Atlantic salmon: cloning, functional characterisation, and tissue distribution of an Elovl4 elongase","volume":"159","author":"Carmona-Anto\u00f1anzas","year":"2011","journal-title":"Comp. Biochem. Physiol. B Biochem. Mol. Biol."},{"key":"10.1016\/j.jlr.2024.100518_bib16","doi-asserted-by":"crossref","first-page":"W171","DOI":"10.1093\/nar\/gkz365","article-title":"CHOPCHOP v3: expanding the CRISPR web toolbox beyond genome editing","volume":"47","author":"Labun","year":"2019","journal-title":"Nucleic Acids Res."},{"key":"10.1016\/j.jlr.2024.100518_bib17","series-title":"An Accessible Protocol for the Generation of CRISPR-Cas9 Knockouts Using INDELs in Zebrafish. Methods in Molecular Biology. 1920","first-page":"377","author":"Moravec","year":"2019"},{"key":"10.1016\/j.jlr.2024.100518_bib18","doi-asserted-by":"crossref","first-page":"26","DOI":"10.1016\/j.ebiom.2016.02.003","article-title":"Targeting tissue lipids in age-related macular degeneration","volume":"5","author":"Apte","year":"2016","journal-title":"EBioMedicine"},{"key":"10.1016\/j.jlr.2024.100518_bib19","doi-asserted-by":"crossref","first-page":"731","DOI":"10.1016\/j.exer.2005.04.012","article-title":"Esterified and unesterified cholesterol in drusen and basal deposits of eyes with age-related maculopathy","volume":"81","author":"Curcio","year":"2005","journal-title":"Exp. Eye Res."},{"key":"10.1016\/j.jlr.2024.100518_bib20","doi-asserted-by":"crossref","first-page":"2214","DOI":"10.3390\/cells9102214","article-title":"Progressive photoreceptor dysfunction and age-related macular degeneration-like features in rp1l1 mutant zebrafish","volume":"9","author":"Noel","year":"2020","journal-title":"Cells"},{"key":"10.1016\/j.jlr.2024.100518_bib21","doi-asserted-by":"crossref","first-page":"11469","DOI":"10.1074\/jbc.M111.256073","article-title":"Essential role of ELOVL4 protein in very long chain fatty acid synthesis and retinal function","volume":"287","author":"Harkewicz","year":"2012","journal-title":"J.\u00a0Biol. Chem."},{"key":"10.1016\/j.jlr.2024.100518_bib22","doi-asserted-by":"crossref","DOI":"10.7554\/eLife.55637","article-title":"Start codon disruption with CRISPR\/Cas9 prevents murine Fuchs\u2019 endothelial corneal dystrophy","volume":"10","author":"Uehara","year":"2021","journal-title":"Elife"},{"key":"10.1016\/j.jlr.2024.100518_bib23","doi-asserted-by":"crossref","DOI":"10.1002\/rcm.9527","article-title":"PE, or not PE, that is the question: the case of overlooked lyso-N-acylphosphatidylethanolamines","volume":"37","author":"Ventura","year":"2023","journal-title":"Rapid Commun. Mass Spectrom."},{"key":"10.1016\/j.jlr.2024.100518_bib24","doi-asserted-by":"crossref","first-page":"273","DOI":"10.1016\/0167-4838(82)90361-2","article-title":"The effect of phospholipid structure on the thermal-stability of rhodopsin","volume":"707","author":"Fischer","year":"1982","journal-title":"Biochim. Biophys. Acta"},{"key":"10.1016\/j.jlr.2024.100518_bib25","doi-asserted-by":"crossref","first-page":"2937","DOI":"10.1038\/s41598-017-02822-w","article-title":"Statistical analysis of zebrafish locomotor behaviour by generalized linear mixed models","volume":"7","author":"Liu","year":"2017","journal-title":"Sci. Rep."},{"key":"10.1016\/j.jlr.2024.100518_bib26","doi-asserted-by":"crossref","first-page":"1185","DOI":"10.3390\/ijms18061185","article-title":"Utilizing zebrafish visual behaviors in drug screening for retinal degeneration","volume":"18","author":"Ganzen","year":"2017","journal-title":"Int. J. Mol. Sci."},{"key":"10.1016\/j.jlr.2024.100518_bib27","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0121330","article-title":"Transducin duplicates in the zebrafish retina and pineal complex: differential specialisation after the teleost tetraploidisation","volume":"10","author":"Lagman","year":"2015","journal-title":"PLoS One"},{"key":"10.1016\/j.jlr.2024.100518_bib28","doi-asserted-by":"crossref","first-page":"10112","DOI":"10.1038\/s41598-022-14361-0","article-title":"Determination of very long-chain polyunsaturated fatty acids from 24 to 44 carbons in eye, brain and gonads of wild and cultured gilthead sea bream (Sparus aurata)","volume":"12","author":"Serrano","year":"2022","journal-title":"Sci. Rep."},{"key":"10.1016\/j.jlr.2024.100518_bib29","doi-asserted-by":"crossref","DOI":"10.1172\/jci.insight.152109","article-title":"Diminished retinal complex lipid synthesis and impaired fatty acid \u03b2-oxidation associated with human diabetic retinopathy","volume":"6","author":"Fort","year":"2021","journal-title":"JCI Insight"},{"key":"10.1016\/j.jlr.2024.100518_bib30","doi-asserted-by":"crossref","DOI":"10.1093\/gigascience\/giz061","article-title":"LION\/web: a web-based ontology enrichment tool for lipidomic data analysis","volume":"8","author":"Molenaar","year":"2019","journal-title":"Gigascience"},{"key":"10.1016\/j.jlr.2024.100518_bib31","doi-asserted-by":"crossref","first-page":"2583","DOI":"10.3390\/cells9122583","article-title":"Elovl2 is required for robust visual function in zebrafish","volume":"9","author":"Dasyani","year":"2020","journal-title":"Cells"},{"key":"10.1016\/j.jlr.2024.100518_bib32","doi-asserted-by":"crossref","first-page":"346","DOI":"10.1016\/j.exer.2018.09.023","article-title":"The impact of lipids, lipid oxidation, and inflammation on AMD, and the potential role of miRNAs on lipid metabolism in the RPE","volume":"181","author":"Jun","year":"2019","journal-title":"Exp. Eye Res."},{"key":"10.1016\/j.jlr.2024.100518_bib33","doi-asserted-by":"crossref","first-page":"869","DOI":"10.1016\/0014-4835(92)90013-I","article-title":"Effect of phytanic acid on cultured retinal-pigment epithelium - an invitro model for refsums disease","volume":"55","author":"Bernstein","year":"1992","journal-title":"Exp. Eye Res."},{"key":"10.1016\/j.jlr.2024.100518_bib34","doi-asserted-by":"crossref","first-page":"333","DOI":"10.3389\/fcell.2019.00333","article-title":"Slc7a14 is indispensable in zebrafish retinas","volume":"7","author":"Zhuang","year":"2019","journal-title":"Front. Cell Dev. Biol."},{"key":"10.1016\/j.jlr.2024.100518_bib35","doi-asserted-by":"crossref","first-page":"257","DOI":"10.1001\/archopht.124.2.257","article-title":"Association of adipose and red blood cell lipids with severity of dominant Stargardt macular dystrophy (STGD3) secondary to an ELOVL4 mutation","volume":"124","author":"Hubbard","year":"2006","journal-title":"Arch. Ophthalmol."},{"key":"10.1016\/j.jlr.2024.100518_bib36","doi-asserted-by":"crossref","first-page":"307","DOI":"10.1080\/13816810.2018.1430240","article-title":"Long-term follow-up of autosomal dominant Stargardt macular dystrophy (STGD3) subjects enrolled in a fish oil supplement interventional trial","volume":"39","author":"Choi","year":"2018","journal-title":"Ophthalmic Genet."},{"key":"10.1016\/j.jlr.2024.100518_bib41","doi-asserted-by":"crossref","first-page":"191","DOI":"10.1016\/j.chroma.2013.07.103","article-title":"Comprehensive and sensitive quantification of long-chain and very long-chain polyunsaturated fatty acids in small samples of human and mouse retina","volume":"1307","author":"Liu","year":"2013","journal-title":"J. Chromatogr. A"},{"key":"10.1016\/j.jlr.2024.100518_bib42","doi-asserted-by":"crossref","first-page":"331","DOI":"10.1186\/s12859-017-1744-3","article-title":"LipidMatch: an automated workflow for rule-based lipid identification using untargeted high-resolution tandem mass spectrometry data","volume":"18","author":"Koelmel","year":"2017","journal-title":"BMC Bioinformatics"},{"key":"10.1016\/j.jlr.2024.100518_bib43","doi-asserted-by":"crossref","first-page":"W251","DOI":"10.1093\/nar\/gkv380","article-title":"MetaboAnalyst 3.0--making metabolomics more meaningful","volume":"43","author":"Xia","year":"2015","journal-title":"Nucleic Acids Res."},{"key":"10.1016\/j.jlr.2024.100518_bib44","doi-asserted-by":"crossref","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"},{"key":"10.1016\/j.jlr.2024.100518_bib45","doi-asserted-by":"crossref","first-page":"2841","DOI":"10.1167\/iovs.02-0991","article-title":"Evolutionarily conserved ELOVL4 gene expression in the vertebrate retina","volume":"44","author":"Lagali","year":"2003","journal-title":"Invest. Ophthalmol. Vis. Sci."},{"key":"10.1016\/j.jlr.2024.100518_bib46","doi-asserted-by":"crossref","first-page":"W695","DOI":"10.1093\/nar\/gkq313","article-title":"A new bioinformatics analysis tools framework at EMBL-EBI","volume":"38","author":"Goujon","year":"2010","journal-title":"Nucleic Acids Res."},{"key":"10.1016\/j.jlr.2024.100518_bib47","doi-asserted-by":"crossref","first-page":"476","DOI":"10.1016\/j.plipres.2010.07.002","article-title":"Defective lipid transport and biosynthesis in recessive and dominant Stargardt macular degeneration","volume":"49","author":"Molday","year":"2010","journal-title":"Prog. Lipid Res."},{"key":"10.1016\/j.jlr.2024.100518_bib48","doi-asserted-by":"crossref","first-page":"378","DOI":"10.1093\/bioinformatics\/14.4.378","article-title":"SOSUI: classification and secondary structure prediction system for membrane proteins","volume":"14","author":"Hirokawa","year":"1998","journal-title":"Bioinformatics"},{"key":"10.1016\/j.jlr.2024.100518_bib49","doi-asserted-by":"crossref","first-page":"240","DOI":"10.1016\/j.aquatox.2015.11.029","article-title":"Stability evaluation of reference genes for real-time PCR in zebrafish (Danio rerio) exposed to cadmium chloride and subsequently infected by bacteria Aeromonas hydrophila","volume":"170","author":"Lang","year":"2016","journal-title":"Aquat. Toxicol."},{"key":"10.1016\/j.jlr.2024.100518_bib50","article-title":"Molecular and functional characterisation of a putative elovl4 gene and its expression in response to dietary fatty acid profile in Atlantic bluefin tuna (Thunnus thynnus)","volume":"240","author":"Betancor","year":"2020","journal-title":"Comp. Biochem. Phys. B\u00a0Biochem. Mol. Biol."}],"container-title":["Journal of Lipid Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0022227524000233?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0022227524000233?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,9,19]],"date-time":"2025-09-19T18:34:57Z","timestamp":1758306897000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0022227524000233"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,3]]},"references-count":46,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2024,3]]}},"alternative-id":["S0022227524000233"],"URL":"https:\/\/doi.org\/10.1016\/j.jlr.2024.100518","relation":{},"ISSN":["0022-2275"],"issn-type":[{"value":"0022-2275","type":"print"}],"subject":[],"published":{"date-parts":[[2024,3]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Elovl4b knockout\u00a0zebrafish as a model for ocular very-long-chain PUFA deficiency","name":"articletitle","label":"Article Title"},{"value":"Journal of Lipid Research","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.jlr.2024.100518","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2024 The Authors. Published by Elsevier Inc on behalf of American Society for Biochemistry and Molecular Biology.","name":"copyright","label":"Copyright"}],"article-number":"100518"}}