{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,22]],"date-time":"2026-09-22T03:36:01Z","timestamp":1790048161235,"version":"4.0.1"},"reference-count":41,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2025,9,1]],"date-time":"2025-09-01T00:00:00Z","timestamp":1756684800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2025,9,1]],"date-time":"2025-09-01T00:00:00Z","timestamp":1756684800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2025,7,15]],"date-time":"2025-07-15T00:00:00Z","timestamp":1752537600000},"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":["EY010609"],"award-info":[{"award-number":["EY010609"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["EY034671"],"award-info":[{"award-number":["EY034671"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000053","name":"National Eye Institute","doi-asserted-by":"publisher","award":["EY033872"],"award-info":[{"award-number":["EY033872"]}],"id":[{"id":"10.13039\/100000053","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100001116","name":"Foundation Fighting Blindness","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\/501100000262","name":"Foundation Fighting Blindness","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000262","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Redox Biology"],"published-print":{"date-parts":[[2025,9]]},"DOI":"10.1016\/j.redox.2025.103772","type":"journal-article","created":{"date-parts":[[2025,7,16]],"date-time":"2025-07-16T07:40:17Z","timestamp":1752651617000},"page":"103772","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":3,"special_numbering":"C","title":["Developmental riboflavin deficiency results in structural and functional changes in the neural retina and RPE"],"prefix":"10.1016","volume":"85","author":[{"given":"Xue","family":"Zhao","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mustafa S.","family":"Makia","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Muna I.","family":"Naash","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4914-350X","authenticated-orcid":false,"given":"Muayyad R.","family":"Al-Ubaidi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"1\u20132","key":"10.1016\/j.redox.2025.103772_bib1","doi-asserted-by":"crossref","first-page":"94","DOI":"10.1016\/j.cbi.2006.04.014","article-title":"Mitochondrial function and toxicity: role of the B vitamin family on mitochondrial energy metabolism","volume":"163","author":"Depeint","year":"2006","journal-title":"Chem. Biol. Interact."},{"issue":"3","key":"10.1016\/j.redox.2025.103772_bib2","doi-asserted-by":"crossref","first-page":"308","DOI":"10.1111\/jns.12587","article-title":"Benefit of high-dose oral riboflavin therapy in riboflavin transporter deficiency","volume":"28","author":"Fennessy","year":"2023","journal-title":"J. Peripher. Nerv. Syst."},{"issue":"6","key":"10.1016\/j.redox.2025.103772_bib3","doi-asserted-by":"crossref","first-page":"943","DOI":"10.1007\/s10545-012-9513-y","article-title":"Impaired riboflavin transport due to missense mutations in SLC52A2 causes Brown-Vialetto-Van laere syndrome","volume":"35","author":"Haack","year":"2012","journal-title":"J. Inherit. Metab. Dis."},{"issue":"6","key":"10.1016\/j.redox.2025.103772_bib4","doi-asserted-by":"crossref","first-page":"1352","DOI":"10.1093\/ajcn\/77.6.1352","article-title":"Riboflavin (vitamin B-2) and health","volume":"77","author":"Powers","year":"2003","journal-title":"Am. J. Clin. Nutr."},{"issue":"3","key":"10.1016\/j.redox.2025.103772_bib5","doi-asserted-by":"crossref","DOI":"10.3390\/ijms21030950","article-title":"Riboflavin: the health benefits of a forgotten natural vitamin","volume":"21","author":"Suwannasom","year":"2020","journal-title":"Int. J. Mol. Sci."},{"issue":"17","key":"10.1016\/j.redox.2025.103772_bib6","doi-asserted-by":"crossref","first-page":"3650","DOI":"10.1080\/10408398.2016.1145104","article-title":"Riboflavin and health: a review of recent human research","volume":"57","author":"Thakur","year":"2017","journal-title":"Crit. Rev. Food Sci. Nutr."},{"issue":"8","key":"10.1016\/j.redox.2025.103772_bib7","doi-asserted-by":"crossref","DOI":"10.3390\/nu11081948","article-title":"Consumption of milk and alternatives and their contribution to nutrient intakes among Canadian adults: evidence from the 2015 Canadian community health survey-nutrition","volume":"11","author":"Auclair","year":"2019","journal-title":"Nutrients"},{"key":"10.1016\/j.redox.2025.103772_bib41","series-title":"StatPearls","article-title":"Vitamin B2 (riboflavin)","author":"Peechakara","year":"2024"},{"issue":"6","key":"10.1016\/j.redox.2025.103772_bib8","doi-asserted-by":"crossref","first-page":"380","DOI":"10.1016\/j.dmpk.2019.08.002","article-title":"Riboflavin transport mediated by riboflavin transporters (RFVTs\/SLC52A) at the rat outer blood-retinal barrier","volume":"34","author":"Kubo","year":"2019","journal-title":"Drug Metabol. Pharmacokinet."},{"issue":"11","key":"10.1016\/j.redox.2025.103772_bib9","doi-asserted-by":"crossref","DOI":"10.3390\/ijms21113847","article-title":"Riboflavin deficiency-implications for general human health and inborn errors of metabolism","volume":"21","author":"Mosegaard","year":"2020","journal-title":"Int. J. Mol. Sci."},{"key":"10.1016\/j.redox.2025.103772_bib10","first-page":"182","article-title":"Riboflavin metabolism--relevance to human nutrition","volume":"108","author":"Lakshmi","year":"1998","journal-title":"Indian J. Med. Res."},{"issue":"4","key":"10.1016\/j.redox.2025.103772_bib11","doi-asserted-by":"crossref","first-page":"605","DOI":"10.1016\/0014-4835(92)90139-J","article-title":"Flavin levels in the rat retina","volume":"54","author":"Batey","year":"1992","journal-title":"Exp. Eye Res."},{"issue":"1","key":"10.1016\/j.redox.2025.103772_bib12","doi-asserted-by":"crossref","first-page":"164","DOI":"10.1016\/0003-2697(90)90546-L","article-title":"Identification of FAD, FMN, and riboflavin in the retina by microextraction and high-performance liquid chromatography","volume":"188","author":"Batey","year":"1990","journal-title":"Anal. Biochem."},{"issue":"8","key":"10.1016\/j.redox.2025.103772_bib13","doi-asserted-by":"crossref","first-page":"5041","DOI":"10.1074\/jbc.M114.624189","article-title":"Retbindin is an extracellular riboflavin-binding protein found at the photoreceptor\/retinal pigment epithelium interface","volume":"290","author":"Kelley","year":"2015","journal-title":"J. Biol. Chem."},{"issue":"51","key":"10.1016\/j.redox.2025.103772_bib14","doi-asserted-by":"crossref","first-page":"21023","DOI":"10.1074\/jbc.M117.785105","article-title":"Ablation of the riboflavin-binding protein retbindin reduces flavin levels and leads to progressive and dose-dependent degeneration of rods and cones","volume":"292","author":"Kelley","year":"2017","journal-title":"J. Biol. Chem."},{"issue":"21","key":"10.1016\/j.redox.2025.103772_bib15","doi-asserted-by":"crossref","DOI":"10.3390\/ijms21218083","article-title":"Retbindin: a riboflavin binding protein, is critical for photoreceptor homeostasis and survival in models of retinal degeneration","volume":"21","author":"Genc","year":"2020","journal-title":"Int. J. Mol. Sci."},{"issue":"6","key":"10.1016\/j.redox.2025.103772_bib16","doi-asserted-by":"crossref","DOI":"10.1073\/pnas.2018956118","article-title":"Absence of retbindin blocks glycolytic flux, disrupts metabolic homeostasis, and leads to photoreceptor degeneration","volume":"118","author":"Sinha","year":"2021","journal-title":"Proc. Natl. Acad. Sci. U. S. A."},{"key":"10.1016\/j.redox.2025.103772_bib17","doi-asserted-by":"crossref","first-page":"101","DOI":"10.1146\/annurev-nutr-061121-084407","article-title":"Causes and clinical sequelae of riboflavin deficiency","volume":"43","author":"McNulty","year":"2023","journal-title":"Annu. Rev. Nutr."},{"issue":"4","key":"10.1016\/j.redox.2025.103772_bib18","doi-asserted-by":"crossref","first-page":"344","DOI":"10.2105\/AJPH.31.4.344","article-title":"Clinical manifestations of ariboflavinosis","volume":"31","author":"Sydenstricker","year":"1941","journal-title":"Am. J. Public Health Nation's Health"},{"key":"10.1016\/j.redox.2025.103772_bib19","doi-asserted-by":"crossref","DOI":"10.1016\/j.pharmthera.2021.108023","article-title":"Recent advances in riboflavin transporter RFVT and its genetic disease","volume":"233","author":"Jin","year":"2022","journal-title":"Pharmacol. Therapeut."},{"key":"10.1016\/j.redox.2025.103772_bib20","doi-asserted-by":"crossref","DOI":"10.1016\/j.redox.2022.102375","article-title":"Riboflavin deficiency leads to irreversible cellular changes in the RPE and disrupts retinal function through alterations in cellular metabolic homeostasis","volume":"54","author":"Sinha","year":"2022","journal-title":"Redox Biol."},{"key":"10.1016\/j.redox.2025.103772_bib21","doi-asserted-by":"crossref","DOI":"10.12659\/AJCR.940439","article-title":"A case report of riboflavin treatment and cochlear implants in a 4-Year-Old girl with progressive hearing loss and delayed speech development: brown-vialetto-van laere syndrome","volume":"24","author":"Piecuch","year":"2023","journal-title":"Am J Case Rep"},{"issue":"12","key":"10.1016\/j.redox.2025.103772_bib22","doi-asserted-by":"crossref","DOI":"10.3390\/brainsci12121605","article-title":"Two rare cases of long surviving riboflavin transporter deficiency with Co-Existing adenosine monophosphate deaminase (AMP) deficiency","volume":"12","author":"Zhang","year":"2022","journal-title":"Brain Sci."},{"issue":"4","key":"10.1016\/j.redox.2025.103772_bib23","doi-asserted-by":"crossref","first-page":"598","DOI":"10.1002\/jimd.12053","article-title":"An update on the genetics, clinical presentation, and pathomechanisms of human riboflavin transporter deficiency","volume":"42","author":"O'Callaghan","year":"2019","journal-title":"J. Inherit. Metab. Dis."},{"key":"10.1016\/j.redox.2025.103772_bib40","series-title":"GeneReviews((R))","article-title":"Riboflavin transporter deficiency","author":"Cali","year":"1993"},{"issue":"1","key":"10.1016\/j.redox.2025.103772_bib24","doi-asserted-by":"crossref","DOI":"10.1038\/s41598-020-75601-9","article-title":"Effect of riboflavin deficiency on development of the cerebral cortex in Slc52a3 knockout mice","volume":"10","author":"Jin","year":"2020","journal-title":"Sci. Rep."},{"key":"10.1016\/j.redox.2025.103772_bib25","doi-asserted-by":"crossref","DOI":"10.1038\/srep27557","article-title":"Disruption of Slc52a3 gene causes neonatal lethality with riboflavin deficiency in mice","volume":"6","author":"Yoshimatsu","year":"2016","journal-title":"Sci. Rep."},{"issue":"2","key":"10.1016\/j.redox.2025.103772_bib26","doi-asserted-by":"crossref","first-page":"283","DOI":"10.1248\/bpb.b20-00751","article-title":"Complete deletion of Slc52a2 causes embryonic lethality in mice","volume":"44","author":"Jin","year":"2021","journal-title":"Biol. Pharm. Bull."},{"key":"10.1016\/j.redox.2025.103772_bib27","doi-asserted-by":"crossref","first-page":"123","DOI":"10.1016\/j.jnutbio.2018.09.003","article-title":"Flavin homeostasis in the mouse retina during aging and degeneration","volume":"62","author":"Sinha","year":"2018","journal-title":"J. Nutr. Biochem."},{"issue":"4","key":"10.1016\/j.redox.2025.103772_bib28","doi-asserted-by":"crossref","DOI":"10.1016\/j.isci.2020.101004","article-title":"The symbiotic relationship between the neural retina and retinal pigment epithelium is supported by utilizing differential metabolic pathways","volume":"23","author":"Sinha","year":"2020","journal-title":"iScience"},{"issue":"6","key":"10.1016\/j.redox.2025.103772_bib29","doi-asserted-by":"crossref","first-page":"17","DOI":"10.1167\/iovs.61.6.17","article-title":"Elimination of a retinal riboflavin binding protein exacerbates degeneration in a model of cone-rod dystrophy","volume":"61","author":"Genc","year":"2020","journal-title":"Investig. Ophthalmol. Vis. Sci."},{"issue":"3","key":"10.1016\/j.redox.2025.103772_bib30","doi-asserted-by":"crossref","first-page":"436","DOI":"10.1111\/acel.12447","article-title":"Retinal pigment epithelial cell multinucleation in the aging eye - a mechanism to repair damage and maintain homoeostasis","volume":"15","author":"Chen","year":"2016","journal-title":"Aging Cell"},{"issue":"9","key":"10.1016\/j.redox.2025.103772_bib31","doi-asserted-by":"crossref","DOI":"10.3390\/cells11091552","article-title":"Multinucleated retinal pigment epithelial cells adapt to vision and exhibit increased DNA damage response","volume":"11","author":"Ke","year":"2022","journal-title":"Cells"},{"issue":"12","key":"10.1016\/j.redox.2025.103772_bib32","doi-asserted-by":"crossref","DOI":"10.3390\/antiox9121252","article-title":"Mitochondrial abnormalities in induced pluripotent stem cells-derived motor neurons from patients with riboflavin transporter deficiency","volume":"9","author":"Colasuonno","year":"2020","journal-title":"Antioxidants"},{"issue":"5","key":"10.1016\/j.redox.2025.103772_bib33","doi-asserted-by":"crossref","DOI":"10.3390\/ijms26051967","article-title":"Recent advances on the role of B vitamins in cancer prevention and progression","volume":"26","author":"Frost","year":"2025","journal-title":"Int. J. Mol. Sci."},{"issue":"2","key":"10.1016\/j.redox.2025.103772_bib34","doi-asserted-by":"crossref","first-page":"89","DOI":"10.7812\/TPP\/21.204","article-title":"B vitamins: functions and uses in medicine","volume":"26","author":"Hanna","year":"2022","journal-title":"Perm. J."},{"issue":"1","key":"10.1016\/j.redox.2025.103772_bib35","doi-asserted-by":"crossref","first-page":"131","DOI":"10.1042\/BST20180420","article-title":"The chemistry of the vitamin B3 metabolome","volume":"47","author":"Makarov","year":"2019","journal-title":"Biochem. Soc. Trans."},{"key":"10.1016\/j.redox.2025.103772_bib36","doi-asserted-by":"crossref","first-page":"50","DOI":"10.1186\/s12934-015-0235-3","article-title":"Acthi, a thiazole biosynthesis enzyme, is essential for thiamine biosynthesis and CPC production in Acremonium chrysogenum","volume":"14","author":"Liu","year":"2015","journal-title":"Microb. Cell Fact."},{"issue":"21","key":"10.1016\/j.redox.2025.103772_bib37","doi-asserted-by":"crossref","first-page":"12929","DOI":"10.1074\/jbc.273.21.12929","article-title":"Activation of thiamin diphosphate and FAD in the phosphatedependent pyruvate oxidase from Lactobacillus plantarum","volume":"273","author":"Tittmann","year":"1998","journal-title":"J. Biol. Chem."},{"issue":"2","key":"10.1016\/j.redox.2025.103772_bib38","doi-asserted-by":"crossref","DOI":"10.3390\/biomedicines11020310","article-title":"Retinal pigment epithelium cell development: extrapolating basic biology to stem cell research","volume":"11","author":"Gupta","year":"2023","journal-title":"Biomedicines"},{"issue":"11","key":"10.1016\/j.redox.2025.103772_bib39","doi-asserted-by":"crossref","DOI":"10.1016\/j.isci.2020.101672","article-title":"Metabolic features of mouse and human retinas: rods versus cones, macula versus periphery, retina versus RPE","volume":"23","author":"Li","year":"2020","journal-title":"iScience"}],"container-title":["Redox Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S221323172500285X?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S221323172500285X?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,5,22]],"date-time":"2026-05-22T10:48:20Z","timestamp":1779446900000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S221323172500285X"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,9]]},"references-count":41,"alternative-id":["S221323172500285X"],"URL":"https:\/\/doi.org\/10.1016\/j.redox.2025.103772","relation":{},"ISSN":["2213-2317"],"issn-type":[{"value":"2213-2317","type":"print"}],"subject":[],"published":{"date-parts":[[2025,9]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Developmental riboflavin deficiency results in structural and functional changes in the neural retina and RPE","name":"articletitle","label":"Article Title"},{"value":"Redox Biology","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.redox.2025.103772","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2025 The Authors. Published by Elsevier B.V.","name":"copyright","label":"Copyright"}],"article-number":"103772"}}