{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,29]],"date-time":"2026-08-29T07:53:58Z","timestamp":1787990038799,"version":"build-2784847793"},"update-to":[{"DOI":"10.1371\/journal.pbio.3002402","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2023,12,14]],"date-time":"2023-12-14T00:00:00Z","timestamp":1702512000000}}],"reference-count":85,"publisher":"Public Library of Science (PLoS)","issue":"12","license":[{"start":{"date-parts":[[2023,12,4]],"date-time":"2023-12-04T00:00:00Z","timestamp":1701648000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/publicdomain\/zero\/1.0\/"}],"funder":[{"DOI":"10.13039\/100005156","name":"Alexander von Humboldt-Stiftung","doi-asserted-by":"publisher","award":["Sofja Kovalewskaja"],"award-info":[{"award-number":["Sofja Kovalewskaja"]}],"id":[{"id":"10.13039\/100005156","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"publisher","award":["MA 6139\/3-1"],"award-info":[{"award-number":["MA 6139\/3-1"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"publisher","award":["MA 6139\/5-1"],"award-info":[{"award-number":["MA 6139\/5-1"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004350","name":"Studienstiftung des Deutschen Volkes","doi-asserted-by":"publisher","award":["PhD Award"],"award-info":[{"award-number":["PhD Award"]}],"id":[{"id":"10.13039\/501100004350","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.plosbiology.org"],"crossmark-restriction":false},"short-container-title":["PLoS Biol"],"abstract":"<jats:p>Vision impairment places a serious burden on the aging society, affecting the lives of millions of people. Many retinal diseases are of genetic origin, of which over 50% are due to mutations in cilia-associated genes. Most research on retinal degeneration has focused on the ciliated photoreceptor cells of the retina. However, the contribution of primary cilia in other ocular cell types has largely been ignored. The retinal pigment epithelium (RPE) is a monolayer epithelium at the back of the eye intricately associated with photoreceptors and essential for visual function. It is already known that primary cilia in the RPE are critical for its development and maturation; however, it remains unclear whether this affects RPE function and retinal tissue homeostasis. We generated a conditional knockout mouse model, in which IFT20 is exclusively deleted in the RPE, ablating primary cilia. This leads to defective RPE function, followed by photoreceptor degeneration and, ultimately, vision impairment. Transcriptomic analysis offers insights into mechanisms underlying pathogenic changes, which include transcripts related to epithelial homeostasis, the visual cycle, and phagocytosis. Due to the loss of cilia exclusively in the RPE, this mouse model enables us to tease out the functional role of RPE cilia and their contribution to retinal degeneration, providing a powerful tool for basic and translational research in syndromic and non-syndromic retinal degeneration. Non-ciliary mechanisms of IFT20 in the RPE may also contribute to pathogenesis and cannot be excluded, especially considering the increasing evidence of non-ciliary functions of ciliary proteins.<\/jats:p>","DOI":"10.1371\/journal.pbio.3002402","type":"journal-article","created":{"date-parts":[[2023,12,4]],"date-time":"2023-12-04T18:36:16Z","timestamp":1701714976000},"page":"e3002402","update-policy":"https:\/\/doi.org\/10.1371\/journal.pbio.corrections_policy","source":"Crossref","is-referenced-by-count":16,"title":["Deletion of IFT20 exclusively in the RPE ablates primary cilia and leads to retinal degeneration"],"prefix":"10.1371","volume":"21","author":[{"given":"Viola","family":"Kretschmer","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sandra","family":"Schneider","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter Andreas","family":"Matthiessen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dominik","family":"Reichert","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathan","family":"Hotaling","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gunnar","family":"Glas\u00dfer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ingo","family":"Lieberwirth","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kapil","family":"Bharti","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rossella","family":"De Cegli","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ivan","family":"Conte","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emeline F.","family":"Nandrot","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9477-4036","authenticated-orcid":true,"given":"Helen Louise","family":"May-Simera","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2023,12,4]]},"reference":[{"issue":"1","key":"pbio.3002402.ref001","doi-asserted-by":"crossref","first-page":"7","DOI":"10.1186\/2046-2530-1-7","article-title":"Cilia, Wnt signaling, and the cytoskeleton.","volume":"1","author":"HL May-Simera","year":"2012","journal-title":"Cilia."},{"key":"pbio.3002402.ref002","doi-asserted-by":"crossref","first-page":"144","DOI":"10.1016\/j.preteyeres.2017.05.001","article-title":"Cilia\u2014The sensory antennae in the eye.","volume":"60","author":"H May-Simera","year":"2017","journal-title":"Prog Retin Eye Res"},{"issue":"4","key":"pbio.3002402.ref003","doi-asserted-by":"crossref","first-page":"222","DOI":"10.1038\/nrm3085","article-title":"Ciliogenesis: building the cell\u2019s antenna","volume":"12","author":"H Ishikawa","year":"2011","journal-title":"Nat Rev Mol Cell Biol"},{"issue":"11","key":"pbio.3002402.ref004","doi-asserted-by":"crossref","DOI":"10.3390\/ijms18112272","article-title":"Primary cilium-dependent signaling mechanisms.","volume":"18","author":"R Pala","year":"2017","journal-title":"Int J Mol Sci."},{"issue":"11","key":"pbio.3002402.ref005","doi-asserted-by":"crossref","first-page":"880","DOI":"10.1038\/nrm2278","article-title":"When cilia go bad: cilia defects and ciliopathies","volume":"8","author":"M Fliegauf","year":"2007","journal-title":"Nat Rev Mol Cell Biol"},{"issue":"7","key":"pbio.3002402.ref006","doi-asserted-by":"crossref","first-page":"1039","DOI":"10.1007\/s00467-010-1731-7","article-title":"Ciliopathies: An expanding disease spectrum","volume":"26","author":"AM Waters","year":"2011","journal-title":"Pediatric Nephrology"},{"issue":"1","key":"pbio.3002402.ref007","article-title":"Primary Cilium Mediated Retinal Pigment Epithelium Maturation is Retarded in Ciliopathy Patient Cells","volume":"22","author":"HL May-Simera","year":"2018","journal-title":"Cell Rep"},{"issue":"February","key":"pbio.3002402.ref008","first-page":"1","article-title":"Loss of Ciliary Gene Bbs8 Results in Physiological Defects in the Retinal Pigment Epithelium.","volume":"9","author":"S Schneider","year":"2021","journal-title":"Front Cell Dev Biol"},{"issue":"SUPPL. 1","key":"pbio.3002402.ref009","doi-asserted-by":"crossref","first-page":"2","DOI":"10.1111\/j.1442-9071.2010.02363.x","article-title":"Anatomy and physiology of the human eye: Effects of mucopolysaccharidoses disease on structure and function\u2014a review","volume":"38","author":"CE Willoughby","year":"2010","journal-title":"Clin Exp Ophthalmol"},{"key":"pbio.3002402.ref010","doi-asserted-by":"crossref","first-page":"845","DOI":"10.1152\/physrev.00021.2004","article-title":"The Retinal Pigment Epithelium in Visual Function","volume":"85","author":"O. Strauss","year":"2005","journal-title":"Physiol Rev"},{"issue":"February","key":"pbio.3002402.ref011","doi-asserted-by":"crossref","first-page":"100846","DOI":"10.1016\/j.preteyeres.2020.100846","article-title":"The cell biology of the retinal pigment epithelium","volume":"78","author":"A Lakkaraju","year":"2020","journal-title":"Prog Retin Eye Res"},{"issue":"1","key":"pbio.3002402.ref012","doi-asserted-by":"crossref","first-page":"21","DOI":"10.1111\/j.1755-148X.2010.00772.x","article-title":"The new paradigm: retinal pigment epithelium cells generated from embryonic or induced pluripotent stem cells","volume":"24","author":"K Bharti","year":"2011","journal-title":"Pigment Cell Melanoma Res"},{"issue":"1","key":"pbio.3002402.ref013","doi-asserted-by":"crossref","DOI":"10.1242\/dmm.035642","article-title":"Photoreceptor degeneration in microphthalmia (Mitf) mice: Partial rescue by pigment epithelium-derived factor.","volume":"12","author":"Y Chen","year":"2019","journal-title":"Dis Model Mech."},{"issue":"3","key":"pbio.3002402.ref014","doi-asserted-by":"crossref","first-page":"377","DOI":"10.1083\/jcb.200808137","article-title":"Deletion of IFT20 in the mouse kidney causes misorientation of the mitotic spindle and cystic kidney disease","volume":"183","author":"JA Jonassen","year":"2008","journal-title":"J Cell Biol"},{"issue":"1","key":"pbio.3002402.ref015","doi-asserted-by":"crossref","first-page":"132","DOI":"10.1016\/j.ydbio.2009.06.023","article-title":"Pax6 dosage requirements in iris and ciliary body differentiation","volume":"333","author":"N Davis","year":"2009","journal-title":"Dev Biol"},{"issue":"7","key":"pbio.3002402.ref016","doi-asserted-by":"crossref","first-page":"889","DOI":"10.1242\/jcs.023861","article-title":"Intraflagellar transport at a glance","volume":"122","author":"L Hao","year":"2009","journal-title":"J Cell Sci"},{"issue":"9","key":"pbio.3002402.ref017","doi-asserted-by":"crossref","first-page":"3781","DOI":"10.1091\/mbc.e06-02-0133","article-title":"The Intraflagellar Transport Protein IFT20 Is Associated with the Golgi Complex and Is Required for Cilia Assembly. Margolis B, editor","volume":"17","author":"JA Follit","year":"2006","journal-title":"Mol Biol Cell"},{"issue":"3","key":"pbio.3002402.ref018","doi-asserted-by":"crossref","first-page":"377","DOI":"10.1083\/jcb.200808137","article-title":"Deletion of IFT20 in the mouse kidney causes misorientation of the mitotic spindle and cystic kidney disease","volume":"183","author":"JA Jonassen","year":"2008","journal-title":"J Cell Biol"},{"issue":"4","key":"pbio.3002402.ref019","doi-asserted-by":"crossref","first-page":"757","DOI":"10.1007\/s00018-018-2966-x","article-title":"Bardet\u2013Biedl Syndrome proteins regulate cilia disassembly during tissue maturation","volume":"76","author":"SR Patnaik","year":"2019","journal-title":"Cell Mol Life Sci"},{"issue":"1","key":"pbio.3002402.ref020","doi-asserted-by":"crossref","first-page":"133","DOI":"10.1038\/nn.2467","article-title":"A robust and high-throughput Cre reporting and characterization system for the whole mouse brain","volume":"13","author":"L Madisen","year":"2010","journal-title":"Nat Neurosci"},{"key":"pbio.3002402.ref021","doi-asserted-by":"crossref","first-page":"51","DOI":"10.1016\/j.exer.2014.01.010","article-title":"Understanding photoreceptor outer segment phagocytosis: use and utility of RPE cells in culture","volume":"126","author":"F Mazzoni","year":"2016","journal-title":"Exp Eye Res"},{"issue":"6","key":"pbio.3002402.ref022","doi-asserted-by":"crossref","first-page":"1330","DOI":"10.1242\/dev.086603","article-title":"Conditional knockdown of DNA methyltransferase 1 reveals a key role of retinal pigment epithelium integrity in photoreceptor outer segment morphogenesis","volume":"140","author":"IO Nasonkin","year":"2013","journal-title":"Development"},{"key":"pbio.3002402.ref023","article-title":"Mouse Retinal Organoid Growth and Maintenance in Longer-Term Culture.","author":"M V\u00f6lkner","year":"2023","journal-title":"Front Cell Dev Biol"},{"key":"pbio.3002402.ref024","doi-asserted-by":"crossref","first-page":"44","DOI":"10.1016\/j.preteyeres.2014.06.003","article-title":"Functional architecture of the retina: development and disease.","volume":"42","author":"M Hoon","year":"2014","journal-title":"Prog Retin Eye Res"},{"issue":"3","key":"pbio.3002402.ref025","doi-asserted-by":"crossref","first-page":"650","DOI":"10.1083\/jcb.58.3.650","article-title":"Kinetics of rod outer segment renewal in the developing mouse retina","volume":"58","author":"MM Lavail","year":"1973","journal-title":"J Cell Biol"},{"issue":"19","key":"pbio.3002402.ref026","doi-asserted-by":"crossref","DOI":"10.1073\/pnas.2117553119","article-title":"Single-cell\u2013resolution map of human retinal pigment epithelium helps discover subpopulations with differential disease sensitivity","volume":"119","author":"D Ortolan","year":"2022","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"12","key":"pbio.3002402.ref027","doi-asserted-by":"crossref","first-page":"2468","DOI":"10.1093\/hmg\/ddq129","article-title":"Transcriptome analysis and molecular signature of human retinal pigment epithelium","volume":"19","author":"NV Strunnikova","year":"2010","journal-title":"Hum Mol Genet"},{"issue":"1","key":"pbio.3002402.ref028","first-page":"1","article-title":"BMP-4 enhances epithelial mesenchymal transition and cancer stem cell properties of breast cancer cells via Notch signaling","volume":"9","author":"S Choi","year":"2019","journal-title":"Sci Rep"},{"issue":"12","key":"pbio.3002402.ref029","doi-asserted-by":"crossref","first-page":"9475","DOI":"10.1007\/s13277-015-3681-y","article-title":"BMP-2 induces motility and invasiveness by promoting colon cancer stemness through STAT3 activation.","volume":"36","author":"BR Kim","year":"2015","journal-title":"Tumor Biol."},{"issue":"24","key":"pbio.3002402.ref030","doi-asserted-by":"crossref","first-page":"4315","DOI":"10.1242\/dev.008151","article-title":"Cessation of gastrulation is mediated by suppression of epithelial-mesenchymal transition at the ventral ectodermal ridge","volume":"134","author":"S Ohta","year":"2007","journal-title":"Development"},{"issue":"Iii","key":"pbio.3002402.ref031","first-page":"1","article-title":"KRT6A Promotes EMT and Cancer Stem Cell Transformation in Lung Adenocarcinoma.","volume":"19","author":"B Yang","year":"2020","journal-title":"Technol Cancer Res Treat"},{"key":"pbio.3002402.ref032","doi-asserted-by":"crossref","first-page":"103","DOI":"10.1007\/978-1-4939-7720-8_7","article-title":"Use of Direct Current Electroretinography for Analysis of Retinal Pigment Epithelium Function in Mouse Models.","volume":"1753","author":"M Yu","year":"2018","journal-title":"Methods Mol Biol"},{"issue":"3","key":"pbio.3002402.ref033","doi-asserted-by":"crossref","first-page":"1134","DOI":"10.1152\/jn.00958.2003","article-title":"Light-evoked responses of the mouse retinal pigment epithelium","volume":"91","author":"J Wu","year":"2004","journal-title":"J Neurophysiol"},{"key":"pbio.3002402.ref034","first-page":"13","article-title":"The Cellular Origin of the Fast Oscillation.","author":"ER Griff","year":"1983"},{"issue":"4","key":"pbio.3002402.ref035","doi-asserted-by":"crossref","first-page":"1669","DOI":"10.1152\/jn.1993.70.4.1669","article-title":"Light-evoked modulation of basolateral membrane Cl- conductance in chick retinal pigment epithelium: The light peak and fast oscillation","volume":"70","author":"RP Gallemore","year":"1993","journal-title":"J Neurophysiol"},{"issue":"4","key":"pbio.3002402.ref036","doi-asserted-by":"crossref","first-page":"1256","DOI":"10.1152\/ajpcell.00480.2005","article-title":"Novel role for \u03b1v\u03b25-integrin in retinal adhesion and its diurnal peak","volume":"290","author":"EF Nandrot","year":"2006","journal-title":"Am J Physiol Cell Physiol"},{"key":"pbio.3002402.ref037","first-page":"1","article-title":"Large-scale purification of porcine or bovine photoreceptor outer segments for phagocytosis assays on retinal pigment epithelial cells.","volume":"94","author":"C Parinot","year":"2014","journal-title":"J Vis Exp"},{"issue":"5","key":"pbio.3002402.ref038","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0096494","article-title":"Spectral-domain optical coherence tomography of the rodent eye: highlighting layers of the outer retina using signal averaging and comparison with histology.","volume":"9","author":"A Berger","year":"2014","journal-title":"PLoS ONE"},{"issue":"11","key":"pbio.3002402.ref039","doi-asserted-by":"crossref","first-page":"e0207222","DOI":"10.1371\/journal.pone.0207222","article-title":"Generation of an inducible RPE-specific Cre transgenic-mouse line.","volume":"13","author":"S Schneider","year":"2018","journal-title":"PLoS ONE."},{"issue":"9","key":"pbio.3002402.ref040","doi-asserted-by":"crossref","first-page":"1521","DOI":"10.1007\/s00018-017-2740-5","article-title":"Primary cilia proteins: ciliary and extraciliary sites and functions","volume":"75","author":"K Hua","year":"2018","journal-title":"Cell Mol Life Sci"},{"issue":"5","key":"pbio.3002402.ref041","doi-asserted-by":"crossref","first-page":"462","DOI":"10.1038\/ng1352","article-title":"The Bardet-Biedl protein BBS4 targets cargo to the pericentriolar region and is required for microtubule anchoring and cell cycle progression","volume":"36","author":"JC Kim","year":"2004","journal-title":"Nat Genet"},{"issue":"7","key":"pbio.3002402.ref042","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1371\/journal.pgen.1006936","article-title":"Nuclear\/cytoplasmic transport defects in BBS6 underlie congenital heart disease through perturbation of a chromatin remodeling protein","volume":"13","author":"CA Scott","year":"2017","journal-title":"PLoS Genet"},{"issue":"2","key":"pbio.3002402.ref043","doi-asserted-by":"crossref","first-page":"362","DOI":"10.1242\/jcs.089375","article-title":"Direct role of Bardet-Biedl syndrome proteins in transcriptional regulation","volume":"125","author":"C Gascue","year":"2012","journal-title":"J Cell Sci"},{"key":"pbio.3002402.ref044","doi-asserted-by":"crossref","DOI":"10.3389\/fcell.2020.578239","article-title":"Non-ciliary Roles of IFT Proteins in Cell Division and Polycystic Kidney Diseases.","volume":"8","author":"B Vitre","year":"2020","journal-title":"Front Cell Dev Biol"},{"issue":"4","key":"pbio.3002402.ref045","doi-asserted-by":"crossref","first-page":"739","DOI":"10.1016\/j.bbrc.2020.09.033","article-title":"IFT20 is critical for collagen biosynthesis in craniofacial bone formation","volume":"533","author":"H Yamaguchi","year":"2020","journal-title":"Biochem Biophys Res Commun"},{"issue":"19","key":"pbio.3002402.ref046","doi-asserted-by":"crossref","first-page":"E2589","DOI":"10.1073\/pnas.1519458113","article-title":"Canonical and noncanonical intraflagellar transport regulates craniofacial skeletal development","volume":"113","author":"K Noda","year":"2016","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"1","key":"pbio.3002402.ref047","doi-asserted-by":"crossref","first-page":"89","DOI":"10.1038\/sj.emboj.7600014","article-title":"Collagen XVIII\/endostatin is essential for vision and retinal pigment epithelial function","volume":"23","author":"AG Marneros","year":"2023","journal-title":"EMBO J"},{"issue":"3","key":"pbio.3002402.ref048","doi-asserted-by":"crossref","DOI":"10.1016\/j.celrep.2020.107932","article-title":"Intraflagellar Transport Complex B Proteins Regulate the Hippo Effector Yap1 during Cardiogenesis.","volume":"32","author":"M Peralta","year":"2020","journal-title":"Cell Rep"},{"issue":"17","key":"pbio.3002402.ref049","first-page":"3021","article-title":"Yap and Taz regulate retinal pigment epithelial cell fate","volume":"142","author":"JB Miesfeld","year":"2015","journal-title":"Development"},{"issue":"20","key":"pbio.3002402.ref050","doi-asserted-by":"crossref","first-page":"3373","DOI":"10.1093\/hmg\/ddaa228","article-title":"Nf2 fine-tunes proliferation and tissue alignment during closure of the optic fissure in the embryonic mouse eye","volume":"29","author":"WR Sun","year":"2020","journal-title":"Hum Mol Genet"},{"issue":"9","key":"pbio.3002402.ref051","doi-asserted-by":"crossref","first-page":"3781","DOI":"10.1091\/mbc.e06-02-0133","article-title":"The intraflagellar transport protein IFT20 is associated with the Golgi complex and is required for cilia assembly","volume":"17","author":"JA Follit","year":"2006","journal-title":"Mol Biol Cell"},{"issue":"3","key":"pbio.3002402.ref052","doi-asserted-by":"crossref","first-page":"555","DOI":"10.1242\/dev.113696","article-title":"Ciliary proteins Bbs8 and Ift20 promote planar cell polarity in the cochlea","volume":"142","author":"HL May-Simera","year":"2015","journal-title":"Development"},{"issue":"7","key":"pbio.3002402.ref053","doi-asserted-by":"crossref","first-page":"921","DOI":"10.1091\/mbc.e10-09-0792","article-title":"IFT20 is required for opsin trafficking and photoreceptor outer segment development","volume":"22","author":"BT Keady","year":"2011","journal-title":"Mol Biol Cell"},{"issue":"6","key":"pbio.3002402.ref054","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.1010796","article-title":"IFT74 variants cause skeletal ciliopathy and motile cilia defects in mice and humans.","volume":"19","author":"Z Bakey","year":"2023","journal-title":"PLoS Genet"},{"issue":"11","key":"pbio.3002402.ref055","doi-asserted-by":"crossref","first-page":"2347","DOI":"10.1242\/dev.127.11.2347","article-title":"The Oak Ridge Polycystic Kidney (orpk) disease gene is required for left-right axis determination.","volume":"127","author":"NS Murcia","year":"2000","journal-title":"Development"},{"issue":"6962","key":"pbio.3002402.ref056","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1038\/nature02061","article-title":"Hedgehog signalling in the mouse requires intraflagellar transport proteins","volume":"426","author":"D Huangfu","year":"2003","journal-title":"Nature"},{"key":"pbio.3002402.ref057","doi-asserted-by":"crossref","DOI":"10.3389\/fcell.2021.607121","article-title":"Loss of Ciliary Gene Bbs8 Results in Physiological Defects in the Retinal Pigment Epithelium.","volume":"9","author":"S Schneider","year":"2021","journal-title":"Front Cell Dev Biol"},{"issue":"14","key":"pbio.3002402.ref058","doi-asserted-by":"crossref","first-page":"R575","DOI":"10.1016\/j.cub.2004.07.017","article-title":"Comparative genomics: prediction of the ciliary and basal body proteome","volume":"14","author":"GJ Pazour","year":"2004","journal-title":"Curr Biol"},{"issue":"17","key":"pbio.3002402.ref059","doi-asserted-by":"crossref","first-page":"6943","DOI":"10.1073\/pnas.1221011110","article-title":"Evolution of modular intraflagellar transport from a coatomer-like progenitor","volume":"110","author":"TJP van Dam","year":"2013","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"August","key":"pbio.3002402.ref060","doi-asserted-by":"crossref","first-page":"105877","DOI":"10.1016\/j.biocel.2020.105877","article-title":"The entangled relationship between cilia and actin","volume":"129","author":"L Br\u00fccker","year":"2020","journal-title":"Int J Biochem Cell Biol"},{"issue":"21","key":"pbio.3002402.ref061","doi-asserted-by":"crossref","first-page":"8145","DOI":"10.1073\/pnas.1121101109","article-title":"Diurnal, localized exposure of phosphatidylserine by rod outer segment tips in wild-type but not Itgb5-\/- or Mfge8-\/- mouse retina","volume":"109","author":"L Ruggiero","year":"2012","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"29","key":"pbio.3002402.ref062","doi-asserted-by":"crossref","first-page":"12005","DOI":"10.1073\/pnas.0704756104","article-title":"Essential role for MFG-E8 as ligand for alphavbeta5 integrin in diurnal retinal phagocytosis","volume":"104","author":"EF Nandrot","year":"2007","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"4","key":"pbio.3002402.ref063","doi-asserted-by":"crossref","DOI":"10.1152\/ajpcell.00480.2005","article-title":"Novel role for alphavbeta5-integrin in retinal adhesion and its diurnal peak","volume":"290","author":"EF Nandrot","year":"2006","journal-title":"Am J Physiol Cell Physiol"},{"issue":"3","key":"pbio.3002402.ref064","doi-asserted-by":"crossref","first-page":"1134","DOI":"10.1152\/jn.00958.2003","article-title":"Light-Evoked Responses of the Mouse Retinal Pigment Epithelium","volume":"91","author":"J Wu","year":"2004","journal-title":"J Neurophysiol"},{"issue":"2","key":"pbio.3002402.ref065","doi-asserted-by":"crossref","first-page":"429","DOI":"10.1113\/jphysiol.2003.042341","article-title":"Regulation of inwardly rectifying K+ channels in retinal pigment epithelial cells by intracellular pH","volume":"549","author":"Y Yuan","year":"2003","journal-title":"J Physiol"},{"issue":"2","key":"pbio.3002402.ref066","doi-asserted-by":"crossref","first-page":"423","DOI":"10.1152\/ajpcell.00393.2007","article-title":"Modulation of the Kir7.1 potassium channel by extracellular and intracellular pH","volume":"294","author":"BA Hughes","year":"2008","journal-title":"Am J Physiol Cell Physiol"},{"issue":"2","key":"pbio.3002402.ref067","doi-asserted-by":"crossref","first-page":"213","DOI":"10.1085\/jgp.83.2.213","article-title":"Delayed basal hyperpolarization of cat retinal pigment epithelium and its relation to the fast oscillation of the DC electroretinogram","volume":"83","author":"RA Linsenmeier","year":"1984","journal-title":"J Gen Physiol"},{"issue":"4","key":"pbio.3002402.ref068","doi-asserted-by":"crossref","first-page":"327","DOI":"10.1007\/BF00158922","article-title":"Interactions between the retinal pigment epithelium and the neural retina.","volume":"60","author":"RH Steinberg","year":"1985","journal-title":"Documenta Ophthalmologica."},{"issue":"2","key":"pbio.3002402.ref069","doi-asserted-by":"crossref","first-page":"450","DOI":"10.1016\/j.bbrc.2018.01.079","article-title":"Deficiency of primary cilia in kidney epithelial cells induces epithelial to mesenchymal transition","volume":"496","author":"SJ Han","year":"2018","journal-title":"Biochem Biophys Res Commun"},{"issue":"5","key":"pbio.3002402.ref070","doi-asserted-by":"crossref","first-page":"547","DOI":"10.1007\/s00418-021-01965-2","article-title":"Primary cilia presence and implications in bladder cancer progression and invasiveness","volume":"155","author":"P Iruzubieta","year":"2021","journal-title":"Histochem Cell Biol"},{"issue":"June","key":"pbio.3002402.ref071","first-page":"1","article-title":"Role of Epithelial-Mesenchymal Transition in Retinal Pigment Epithelium Dysfunction.","volume":"8","author":"M Zhou","year":"2020","journal-title":"Front Cell Dev Biol"},{"issue":"3","key":"pbio.3002402.ref072","doi-asserted-by":"crossref","first-page":"e201800212","DOI":"10.26508\/lsa.201800212","article-title":"Loss of PGC-1\u03b1 in RPE induces mesenchymal transition and promotes retinal degeneration","volume":"2","author":"MAB Rosales","year":"2019","journal-title":"Life Sci Alliance"},{"issue":"22","key":"pbio.3002402.ref073","doi-asserted-by":"crossref","first-page":"3147","DOI":"10.1101\/gad.1700108","article-title":"Retinal degeneration triggered by inactivation of PTEN in the retinal pigment epithelium","volume":"22","author":"WK Jin","year":"2008","journal-title":"Genes Dev"},{"issue":"1","key":"pbio.3002402.ref074","doi-asserted-by":"crossref","first-page":"103","DOI":"10.1083\/jcb.200107108","article-title":"The intraflagellar transport protein, IFT88, is essential for vertebrate photoreceptor assembly and maintenance","volume":"157","author":"GJ Pazour","year":"2002","journal-title":"J Cell Biol"},{"issue":"7","key":"pbio.3002402.ref075","doi-asserted-by":"crossref","first-page":"676","DOI":"10.1038\/nmeth.2019","article-title":"Fiji: An open-source platform for biological-image analysis.","volume":"9","author":"J Schindelin","year":"2012","journal-title":"Nat Methods."},{"issue":"12","key":"pbio.3002402.ref076","doi-asserted-by":"crossref","first-page":"1539","DOI":"10.1084\/jem.20041447","article-title":"Loss of synchronized retinal phagocytosis and age-related blindness in mice lacking \u03b1v\u03b25 integrin","volume":"200","author":"EF Nandrot","year":"2004","journal-title":"J Exp Med"},{"issue":"8","key":"pbio.3002402.ref077","doi-asserted-by":"crossref","first-page":"3612","DOI":"10.1167\/iovs.05-1622","article-title":"Confluent monolayers of cultured human fetal retinal pigment epithelium exhibit morphology and physiology of native tissue","volume":"47","author":"A Maminishkis","year":"2006","journal-title":"Invest Ophthalmol Vis Sci"},{"issue":"1","key":"pbio.3002402.ref078","doi-asserted-by":"crossref","first-page":"27","DOI":"10.1109\/83.650848","article-title":"A pyramid approach to subpixel registration based on intensity","volume":"7","author":"P Th\u00e9venaz","year":"1998","journal-title":"IEEE Trans Image Process"},{"issue":"5","key":"pbio.3002402.ref079","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0096494","article-title":"Spectral-domain optical coherence tomography of the rodent eye: Highlighting layers of the outer retina using signal averaging and comparison with histology.","volume":"9","author":"A Berger","year":"2014","journal-title":"PLoS ONE."},{"issue":"2","key":"pbio.3002402.ref080","first-page":"65","article-title":"A simple sequentially rejective multiple test procedure","volume":"6","author":"S. Holm","year":"1979","journal-title":"Scand J Stat"},{"issue":"5","key":"pbio.3002402.ref081","doi-asserted-by":"crossref","first-page":"321","DOI":"10.1023\/A:1022238410461","article-title":"The life and work of Zbyn\u011bk \u0160id\u00e1k (1933\u20131999).","volume":"45","author":"J Seidler","year":"2000","journal-title":"Appl Math"},{"issue":"3","key":"pbio.3002402.ref082","doi-asserted-by":"crossref","first-page":"363","DOI":"10.3758\/BF03202031","article-title":"FORTRAN IV functions for calculating probabilities associated with Dunnett\u2019s test.","volume":"13","author":"WP Dunlap","year":"1981","journal-title":"Behav Res Meth Instrum."},{"key":"pbio.3002402.ref083","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1016\/j.jneumeth.2015.08.007","article-title":"A system to measure the Optokinetic and Optomotor response in mice","volume":"256","author":"F Kretschmer","year":"2015","journal-title":"J Neurosci Methods"},{"issue":"11","key":"pbio.3002402.ref084","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1371\/journal.pone.0078058","article-title":"OMR-arena: Automated measurement and stimulation system to determine mouse visual thresholds based on optomotor responses.","volume":"8","author":"F Kretschmer","year":"2013","journal-title":"PLoS ONE"},{"issue":"1","key":"pbio.3002402.ref085","doi-asserted-by":"crossref","first-page":"300","DOI":"10.1152\/jn.00055.2017","article-title":"Comparison of optomotor and optokinetic reflexes in mice","volume":"118","author":"F Kretschmer","year":"2017","journal-title":"J Neurophysiol"}],"updated-by":[{"DOI":"10.1371\/journal.pbio.3002402","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2023,12,14]],"date-time":"2023-12-14T00:00:00Z","timestamp":1702512000000}}],"container-title":["PLOS Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pbio.3002402","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,12,14]],"date-time":"2023-12-14T19:08:46Z","timestamp":1702580926000},"score":1,"resource":{"primary":{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pbio.3002402"}},"subtitle":[],"editor":[{"given":"Dagmar","family":"Wachten","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2023,12,4]]},"references-count":85,"journal-issue":{"issue":"12","published-online":{"date-parts":[[2023,12,4]]}},"URL":"https:\/\/doi.org\/10.1371\/journal.pbio.3002402","relation":{"new_version":[{"id-type":"doi","id":"10.1371\/journal.pbio.3002402","asserted-by":"object"}]},"ISSN":["1545-7885"],"issn-type":[{"value":"1545-7885","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,12,4]]}}}