{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,7]],"date-time":"2026-07-07T22:16:33Z","timestamp":1783462593213,"version":"3.55.0"},"reference-count":78,"publisher":"Elsevier BV","issue":"2","license":[{"start":{"date-parts":[[2019,9,1]],"date-time":"2019-09-01T00:00:00Z","timestamp":1567296000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2019,9,1]],"date-time":"2019-09-01T00:00:00Z","timestamp":1567296000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2020,9,18]],"date-time":"2020-09-18T00:00:00Z","timestamp":1600387200000},"content-version":"vor","delay-in-days":383,"URL":"http:\/\/www.elsevier.com\/open-access\/userlicense\/1.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01EY020578"],"award-info":[{"award-number":["R01EY020578"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01NS104038"],"award-info":[{"award-number":["R01NS104038"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100012711","name":"Curing Retinal Blindness Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100012711","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001823","name":"Ministerstvo \u0160kolstv\u00ed, Ml\u00e1de\u017ee a T\u011blov\u00fdchovy","doi-asserted-by":"publisher","award":["LO1419"],"award-info":[{"award-number":["LO1419"]}],"id":[{"id":"10.13039\/501100001823","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Developmental Biology"],"published-print":{"date-parts":[[2019,9]]},"DOI":"10.1016\/j.ydbio.2019.05.008","type":"journal-article","created":{"date-parts":[[2019,5,27]],"date-time":"2019-05-27T20:35:16Z","timestamp":1558989316000},"page":"141-154","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":13,"title":["Targeted deletion of Crb1\/Crb2 in the optic vesicle models key features of leber congenital amaurosis 8"],"prefix":"10.1016","volume":"453","author":[{"given":"Seo-Hee","family":"Cho","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ankur","family":"Nahar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ji Hyang","family":"Kim","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew","family":"Lee","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zbynek","family":"Kozmik","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seonhee","family":"Kim","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"key":"10.1016\/j.ydbio.2019.05.008_bib1","doi-asserted-by":"crossref","first-page":"6898","DOI":"10.1167\/iovs.11-7701","article-title":"Human CRB1-associated retinal degeneration: comparison with the rd8 Crb1-mutant mouse model","volume":"52","author":"Aleman","year":"2011","journal-title":"Investig. Ophthalmol. Vis. Sci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib2","doi-asserted-by":"crossref","first-page":"251","DOI":"10.1007\/s13205-017-0878-3","article-title":"Genetic characterization and disease mechanism of retinitis pigmentosa; current scenario","volume":"7","author":"Ali","year":"2017","journal-title":"3 Biotech"},{"key":"10.1016\/j.ydbio.2019.05.008_bib3","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0082532","article-title":"Microarray and morphological analysis of early postnatal CRB2 mutant retinas on a pure C57BL\/6J genetic background","volume":"8","author":"Alves","year":"2013","journal-title":"PLoS One"},{"key":"10.1016\/j.ydbio.2019.05.008_bib4","doi-asserted-by":"crossref","first-page":"3384","DOI":"10.1093\/hmg\/ddu048","article-title":"Targeted ablation of Crb2 in photoreceptor cells induces retinitis pigmentosa","volume":"23","author":"Alves","year":"2014","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib5","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1016\/j.preteyeres.2014.01.001","article-title":"The CRB1 and adherens junction complex proteins in retinal development and maintenance","volume":"40","author":"Alves","year":"2014","journal-title":"Prog. Retin. Eye Res."},{"key":"10.1016\/j.ydbio.2019.05.008_bib6","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1093\/hmg\/dds398","article-title":"Loss of CRB2 in the mouse retina mimics human retinitis pigmentosa due to mutations in the CRB1 gene","volume":"22","author":"Alves","year":"2013","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib7","doi-asserted-by":"crossref","first-page":"614","DOI":"10.1016\/j.bbamem.2007.08.029","article-title":"Polarity complex proteins","volume":"1778","author":"Assemat","year":"2008","journal-title":"Biochim. Biophys. Acta"},{"key":"10.1016\/j.ydbio.2019.05.008_bib8","doi-asserted-by":"crossref","first-page":"1071","DOI":"10.1038\/ejhg.2015.241","article-title":"Comprehensive genotyping reveals RPE65 as the most frequently mutated gene in Leber congenital amaurosis in Denmark","volume":"24","author":"Astuti","year":"2016","journal-title":"Eur. J. Hum. Genet. : EJHG"},{"key":"10.1016\/j.ydbio.2019.05.008_bib9","doi-asserted-by":"crossref","first-page":"306","DOI":"10.1002\/humu.21653","article-title":"CRB1 mutations in inherited retinal dystrophies","volume":"33","author":"Bujakowska","year":"2012","journal-title":"Hum. Mutat."},{"key":"10.1016\/j.ydbio.2019.05.008_bib10","doi-asserted-by":"crossref","first-page":"1201","DOI":"10.1242\/jcs.188599","article-title":"Development and dynamics of cell polarity at a glance","volume":"130","author":"Campanale","year":"2017","journal-title":"J.\u00a0Cell Sci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib11","doi-asserted-by":"crossref","first-page":"112","DOI":"10.12998\/wjcc.v3.i2.112","article-title":"Review and update on the molecular basis of Leber congenital amaurosis","volume":"3","author":"Chacon-Camacho","year":"2015","journal-title":"World J. Clin. Cases"},{"key":"10.1016\/j.ydbio.2019.05.008_bib12","doi-asserted-by":"crossref","first-page":"4974","DOI":"10.1167\/iovs.13-12289","article-title":"Survey of common eye diseases in laboratory mouse strains","volume":"54","author":"Chang","year":"2013","journal-title":"Investig. Ophthalmol. Vis. Sci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib13","doi-asserted-by":"crossref","first-page":"164","DOI":"10.1016\/j.exer.2018.12.018","article-title":"CRB2 mutation causes autosomal recessive retinitis pigmentosa","volume":"180","author":"Chen","year":"2019","journal-title":"Exp. Eye Res."},{"key":"10.1016\/j.ydbio.2019.05.008_bib14","doi-asserted-by":"crossref","first-page":"4351","DOI":"10.1167\/iovs.13-11606","article-title":"Comprehensive mutation analysis by whole-exome sequencing in 41 Chinese families with Leber congenital amaurosis","volume":"54","author":"Chen","year":"2013","journal-title":"Investig. Ophthalmol. Vis. Sci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib15","doi-asserted-by":"crossref","first-page":"32","DOI":"10.1159\/000342159","article-title":"Hyperreflective dots: a new spectral-domain optical coherence tomography entity for follow-up and prognosis in exudative age-related macular degeneration","volume":"229","author":"Coscas","year":"2013","journal-title":"Ophthalmologica. Journal international d'ophtalmologie. International journal of ophthalmology. Zeitschrift fur Augenheilkunde"},{"key":"10.1016\/j.ydbio.2019.05.008_bib16","doi-asserted-by":"crossref","first-page":"7","DOI":"10.1080\/13816810.2016.1275021","article-title":"Leber congenital amaurosis, from darkness to light: an ode to Irene Maumenee","volume":"38","author":"Coussa","year":"2017","journal-title":"Ophthalmic Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib17","doi-asserted-by":"crossref","first-page":"1169","DOI":"10.1093\/hmg\/11.10.1169","article-title":"Molecular genetics of Leber congenital amaurosis","volume":"11","author":"Cremers","year":"2002","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib18","doi-asserted-by":"crossref","first-page":"355","DOI":"10.1002\/humu.20093","article-title":"CRB1 mutation spectrum in inherited retinal dystrophies","volume":"24","author":"den Hollander","year":"2004","journal-title":"Hum. Mutat."},{"key":"10.1016\/j.ydbio.2019.05.008_bib19","doi-asserted-by":"crossref","first-page":"203","DOI":"10.1016\/S0925-4773(01)00568-8","article-title":"Isolation of Crb1, a mouse homologue of Drosophila crumbs, and analysis of its expression pattern in eye and brain","volume":"110","author":"den Hollander","year":"2002","journal-title":"Mech. Dev."},{"key":"10.1016\/j.ydbio.2019.05.008_bib20","doi-asserted-by":"crossref","first-page":"198","DOI":"10.1086\/321263","article-title":"Leber congenital amaurosis and retinitis pigmentosa with Coats-like exudative vasculopathy are associated with mutations in the crumbs homologue 1 (CRB1) gene","volume":"69","author":"den Hollander","year":"2001","journal-title":"Am. J. Hum. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib21","doi-asserted-by":"crossref","first-page":"391","DOI":"10.1016\/j.preteyeres.2008.05.003","article-title":"Leber congenital amaurosis: genes, proteins and disease mechanisms","volume":"27","author":"den Hollander","year":"2008","journal-title":"Prog. Retin. Eye Res."},{"key":"10.1016\/j.ydbio.2019.05.008_bib22","doi-asserted-by":"crossref","first-page":"217","DOI":"10.1038\/13848","article-title":"Mutations in a human homologue of Drosophila crumbs cause retinitis pigmentosa (RP12)","volume":"23","author":"den Hollander","year":"1999","journal-title":"Nat. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib23","doi-asserted-by":"crossref","first-page":"107","DOI":"10.1016\/j.preteyeres.2017.10.004","article-title":"Molecular genetics and emerging therapies for retinitis pigmentosa: basic research and clinical perspectives","volume":"63","author":"Dias","year":"2018","journal-title":"Prog. Retin. Eye Res."},{"key":"10.1016\/j.ydbio.2019.05.008_bib24","doi-asserted-by":"crossref","first-page":"11652","DOI":"10.1038\/s41598-018-30210-5","article-title":"Expression and localization of the polarity protein CRB2 in adult mouse brain: a comparison with the CRB1(rd8) mutant mouse model","volume":"8","author":"Dolon","year":"2018","journal-title":"Sci. Rep."},{"key":"10.1016\/j.ydbio.2019.05.008_bib25","doi-asserted-by":"crossref","first-page":"677","DOI":"10.1017\/S0952523805225166","article-title":"Independent visual threshold measurements in the two eyes of freely moving rats and mice using a virtual-reality optokinetic system","volume":"22","author":"Douglas","year":"2005","journal-title":"Vis. Neurosci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib26","doi-asserted-by":"crossref","first-page":"397","DOI":"10.3109\/08820538.2013.825277","article-title":"CRB1: one gene, many phenotypes","volume":"28","author":"Ehrenberg","year":"2013","journal-title":"Semin. Ophthalmol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib27","doi-asserted-by":"crossref","first-page":"R2","DOI":"10.1093\/hmg\/ddx185","article-title":"Toward an elucidation of the molecular genetics of inherited retinal degenerations","volume":"26","author":"Farrar","year":"2017","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib28","doi-asserted-by":"crossref","first-page":"1502","DOI":"10.1001\/archopht.1985.01050100078023","article-title":"Classification of congenital and early onset retinitis pigmentosa","volume":"103","author":"Foxman","year":"1985","journal-title":"Arch. Ophthalmol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib29","doi-asserted-by":"crossref","first-page":"153","DOI":"10.1258\/0023677011911525","article-title":"A\u00a0simple polymerase chain reaction assay for genotyping the retinal degeneration mutation (Pdeb(rd1)) in FVB\/N-derived transgenic mice","volume":"35","author":"Gimenez","year":"2001","journal-title":"Lab. Anim."},{"key":"10.1016\/j.ydbio.2019.05.008_bib30","doi-asserted-by":"crossref","first-page":"713","DOI":"10.1016\/j.exer.2008.02.005","article-title":"Composition and function of the Crumbs protein complex in the mammalian retina","volume":"86","author":"Gosens","year":"2008","journal-title":"Exp. Eye Res."},{"key":"10.1016\/j.ydbio.2019.05.008_bib31","doi-asserted-by":"crossref","first-page":"433","DOI":"10.1016\/S0021-5155(02)00504-X","article-title":"The a-wave latency in control subjects and patients with retinal diseases","volume":"46","author":"Hamasaki","year":"2002","journal-title":"Jpn. J. Ophthalmol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib32","doi-asserted-by":"crossref","first-page":"811","DOI":"10.1136\/bjo.2010.186882","article-title":"Phenotypic variability in patients with retinal dystrophies due to mutations in CRB1","volume":"95","author":"Henderson","year":"2011","journal-title":"Br. J. Ophthalmol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib33","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1038\/ncb923","article-title":"Direct interaction of two polarity complexes implicated in epithelial tight junction assembly","volume":"5","author":"Hurd","year":"2003","journal-title":"Nat. Cell Biol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib34","doi-asserted-by":"crossref","first-page":"1073","DOI":"10.1093\/hmg\/ddg117","article-title":"Crumbs homolog 1 (CRB1) mutations result in a thick human retina with abnormal lamination","volume":"12","author":"Jacobson","year":"2003","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib35","doi-asserted-by":"crossref","first-page":"687","DOI":"10.1038\/s41431-017-0082-2","article-title":"A\u00a0clinical and molecular characterisation of CRB1-associated maculopathy","volume":"26","author":"Khan","year":"2018","journal-title":"Eur. J. Hum. Genet. : EJHG"},{"key":"10.1016\/j.ydbio.2019.05.008_bib36","doi-asserted-by":"crossref","first-page":"336","DOI":"10.1016\/j.ydbio.2016.09.001","article-title":"Yap is essential for retinal progenitor cell cycle progression and RPE cell fate acquisition in the developing mouse eye","volume":"419","author":"Kim","year":"2016","journal-title":"Dev. Biol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib37","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0063029","article-title":"Generation of mRx-Cre transgenic mouse line for efficient conditional gene deletion in early retinal progenitors","volume":"8","author":"Klimova","year":"2013","journal-title":"PLoS One"},{"key":"10.1016\/j.ydbio.2019.05.008_bib38","doi-asserted-by":"crossref","first-page":"11222","DOI":"10.1523\/JNEUROSCI.4239-09.2010","article-title":"Age-related deterioration of rod vision in mice","volume":"30","author":"Kolesnikov","year":"2010","journal-title":"J.\u00a0Neurosci. : Off. J. Soc. Neurosci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib39","doi-asserted-by":"crossref","first-page":"670","DOI":"10.1093\/hmg\/ddu487","article-title":"Viral-mediated vision rescue of a novel AIPL1 cone-rod dystrophy model","volume":"24","author":"Ku","year":"2015","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib40","doi-asserted-by":"crossref","first-page":"1147","DOI":"10.1136\/bjophthalmol-2016-309975","article-title":"Leber congenital amaurosis\/early-onset severe retinal dystrophy: clinical features, molecular genetics and therapeutic interventions","volume":"101","author":"Kumaran","year":"2017","journal-title":"Br. J. Ophthalmol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib41","doi-asserted-by":"crossref","first-page":"1436","DOI":"10.1038\/ejhg.2016.24","article-title":"Expansion of phenotype and genotypic data in CRB2-related syndrome","volume":"24","author":"Lamont","year":"2016","journal-title":"Eur. J. Hum. Genet. : EJHG"},{"key":"10.1016\/j.ydbio.2019.05.008_bib42","first-page":"2157","article-title":"A\u00a0functional role of the extracellular domain of Crumbs in cell architecture and apicobasal polarity","volume":"126","author":"Letizia","year":"2013","journal-title":"J.\u00a0Cell Sci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib43","doi-asserted-by":"crossref","first-page":"415","DOI":"10.1001\/archopht.119.3.415","article-title":"Mutations in the CRB1 gene cause Leber congenital amaurosis","volume":"119","author":"Lotery","year":"2001","journal-title":"Arch. Ophthalmol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib44","doi-asserted-by":"crossref","first-page":"21","DOI":"10.1016\/S0378111902010843","article-title":"Mammalian Crumbs3 is a small transmembrane protein linked to protein associated with Lin-7 (Pals1)","volume":"302","author":"Makarova","year":"2003","journal-title":"Gene"},{"key":"10.1016\/j.ydbio.2019.05.008_bib45","doi-asserted-by":"crossref","first-page":"164","DOI":"10.1016\/j.exer.2015.11.013","article-title":"Mthfr as a modifier of the retinal phenotype of Crb1(rd8\/rd8) mice","volume":"145","author":"Markand","year":"2016","journal-title":"Exp. Eye Res."},{"key":"10.1016\/j.ydbio.2019.05.008_bib46","doi-asserted-by":"crossref","first-page":"161","DOI":"10.1097\/IAE.0000000000001127","article-title":"Long-TERM FOLLOW-UP OF patients with retinitis pigmentosa type 12 caused BY CRB1 mutations: a severe phenotype with considerable interindividual variability","volume":"37","author":"Mathijssen","year":"2017","journal-title":"Retina"},{"key":"10.1016\/j.ydbio.2019.05.008_bib47","doi-asserted-by":"crossref","first-page":"2921","DOI":"10.1167\/iovs.12-9662","article-title":"The Rd8 mutation of the Crb1 gene is present in vendor lines of C57BL\/6N mice and embryonic stem cells, and confounds ocular induced mutant phenotypes","volume":"53","author":"Mattapallil","year":"2012","journal-title":"Investig. Ophthalmol. Vis. Sci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib48","doi-asserted-by":"crossref","first-page":"2179","DOI":"10.1093\/hmg\/ddg232","article-title":"CRB1 is essential for external limiting membrane integrity and photoreceptor morphogenesis in the mammalian retina","volume":"12","author":"Mehalow","year":"2003","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib49","doi-asserted-by":"crossref","first-page":"828","DOI":"10.1042\/BST0320828","article-title":"Crumbs homologue 1 in polarity and blindness","volume":"32","author":"Meuleman","year":"2004","journal-title":"Biochem. Soc. Trans."},{"key":"10.1016\/j.ydbio.2019.05.008_bib50","doi-asserted-by":"crossref","first-page":"6446","DOI":"10.1093\/hmg\/ddv354","article-title":"Long-term rescue of cone photoreceptor degeneration in retinitis pigmentosa 2 (RP2)-knockout mice by gene replacement therapy","volume":"24","author":"Mookherjee","year":"2015","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib51","doi-asserted-by":"crossref","first-page":"8654","DOI":"10.1038\/s41598-017-09035-1","article-title":"The correlation between CRB1 variants and the clinical severity of Brazilian patients with different inherited retinal dystrophy phenotypes","volume":"7","author":"Motta","year":"2017","journal-title":"Sci. Rep."},{"key":"10.1016\/j.ydbio.2019.05.008_bib52","doi-asserted-by":"crossref","first-page":"14504","DOI":"10.1038\/srep14504","article-title":"CRB2 completes a fully expressed Crumbs complex in the Retinal Pigment Epithelium","volume":"5","author":"Paniagua","year":"2015","journal-title":"Sci. Rep."},{"key":"10.1016\/j.ydbio.2019.05.008_bib53","doi-asserted-by":"crossref","first-page":"10329","DOI":"10.1038\/ncomms10329","article-title":"Yap is required for ependymal integrity and is suppressed in LPA-induced hydrocephalus","volume":"7","author":"Park","year":"2016","journal-title":"Nat. Commun."},{"key":"10.1016\/j.ydbio.2019.05.008_bib54","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.1003976","article-title":"Targeted ablation of CRB1 and CRB2 in retinal progenitor cells mimics Leber congenital amaurosis","volume":"9","author":"Pellissier","year":"2013","journal-title":"PLoS Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib55","doi-asserted-by":"crossref","first-page":"3759","DOI":"10.1093\/hmg\/ddu089","article-title":"CRB2 acts as a modifying factor of CRB1-related retinal dystrophies in mice","volume":"23","author":"Pellissier","year":"2014","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib56","doi-asserted-by":"crossref","first-page":"4611","DOI":"10.1167\/iovs.04-0541","article-title":"Rapid quantification of adult and developing mouse spatial vision using a virtual optomotor system","volume":"45","author":"Prusky","year":"2004","journal-title":"Investig. Ophthalmol. Vis. Sci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib57","doi-asserted-by":"crossref","first-page":"426","DOI":"10.1016\/S0021-5155(02)00505-1","article-title":"Evidence that a-wave latency of the electroretinogram is determined solely by photoreceptors","volume":"46","author":"Qiu","year":"2002","journal-title":"Jpn. J. Ophthalmol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib58","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1093\/hmg\/ddy337","article-title":"Loss of CRB2 in Muller glial cells modifies a CRB1-associated retinitis pigmentosa phenotype into a Leber congenital amaurosis phenotype","volume":"28","author":"Quinn","year":"2019","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib59","doi-asserted-by":"crossref","first-page":"175","DOI":"10.3389\/fnins.2017.00175","article-title":"The CRB1 complex: following the trail of crumbs to a feasible gene therapy strategy","volume":"11","author":"Quinn","year":"2017","journal-title":"Front. Neurosci."},{"issue":"2","key":"10.1016\/j.ydbio.2019.05.008_bib60","doi-asserted-by":"crossref","first-page":"R235","DOI":"10.1093\/hmg\/ddl195","article-title":"Towards understanding CRUMBS function in retinal dystrophies","volume":"15","author":"Richard","year":"2006","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib61","doi-asserted-by":"crossref","first-page":"139","DOI":"10.1038\/75973","article-title":"High-efficiency deleter mice show that FLPe is an alternative to Cre-loxP","volume":"25","author":"Rodriguez","year":"2000","journal-title":"Nat. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib62","doi-asserted-by":"crossref","first-page":"161","DOI":"10.1083\/jcb.200109010","article-title":"The Maguk protein, Pals1, functions as an adapter, linking mammalian homologues of Crumbs and Discs Lost","volume":"157","author":"Roh","year":"2002","journal-title":"J.\u00a0Cell Biol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib63","doi-asserted-by":"crossref","first-page":"388","DOI":"10.1016\/j.ydbio.2004.03.039","article-title":"Genetic analysis of the homeodomain transcription factor Chx10 in the retina using a novel multifunctional BAC transgenic mouse reporter","volume":"271","author":"Rowan","year":"2004","journal-title":"Dev. Biol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib64","doi-asserted-by":"crossref","first-page":"384","DOI":"10.1016\/j.optm.2008.01.026","article-title":"Review and update: current treatment trends for patients with retinitis pigmentosa","volume":"80","author":"Shintani","year":"2009","journal-title":"Optometry"},{"key":"10.1016\/j.ydbio.2019.05.008_bib65","doi-asserted-by":"crossref","first-page":"356","DOI":"10.1016\/j.cell.2009.01.052","article-title":"Nuclear architecture of rod photoreceptor cells adapts to vision in mammalian evolution","volume":"137","author":"Solovei","year":"2009","journal-title":"Cell"},{"key":"10.1016\/j.ydbio.2019.05.008_bib66","doi-asserted-by":"crossref","first-page":"791","DOI":"10.1016\/j.ajo.2007.08.022","article-title":"Leber congenital amaurosis - a model for efficient genetic testing of heterogeneous disorders: LXIV Edward Jackson Memorial Lecture","volume":"144","author":"Stone","year":"2007","journal-title":"Am. J. Ophthalmol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib67","doi-asserted-by":"crossref","first-page":"884","DOI":"10.1016\/j.ophtha.2017.01.047","article-title":"Genotypic and phenotypic characteristics of CRB1-associated retinal dystrophies: a long-term follow-up study","volume":"124","author":"Talib","year":"2017","journal-title":"Ophthalmology"},{"key":"10.1016\/j.ydbio.2019.05.008_bib68","doi-asserted-by":"crossref","first-page":"655","DOI":"10.1146\/annurev-cellbio-092910-154033","article-title":"The apical polarity protein network in Drosophila epithelial cells: regulation of polarity, junctions, morphogenesis, cell growth, and survival","volume":"28","author":"Tepass","year":"2012","journal-title":"Annu. Rev. Cell Dev. Biol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib69","doi-asserted-by":"crossref","first-page":"307","DOI":"10.1038\/nrm3568","article-title":"Sticking together the Crumbs - an unexpected function for an old friend","volume":"14","author":"Thompson","year":"2013","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"10.1016\/j.ydbio.2019.05.008_bib70","doi-asserted-by":"crossref","first-page":"36","DOI":"10.2174\/1874364101509010036","article-title":"The causes of hyperreflective dots in optical coherence tomography excluding diabetic macular edema and retinal venous occlusion section sign","volume":"9","author":"Turgut","year":"2015","journal-title":"Open Ophthalmol. J."},{"key":"10.1016\/j.ydbio.2019.05.008_bib71","doi-asserted-by":"crossref","first-page":"4169","DOI":"10.1242\/jcs.01301","article-title":"Crumbs homologue 1 is required for maintenance of photoreceptor cell polarization and adhesion during light exposure","volume":"117","author":"van de Pavert","year":"2004","journal-title":"J.\u00a0Cell Sci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib72","doi-asserted-by":"crossref","first-page":"564","DOI":"10.1523\/JNEUROSCI.3496-06.2007","article-title":"A\u00a0single amino acid substitution (Cys249Trp) in Crb1 causes retinal degeneration and deregulates expression of pituitary tumor transforming gene Pttg1","volume":"27","author":"van de Pavert","year":"2007","journal-title":"J.\u00a0Neurosci. : Off. J. Soc. Neurosci."},{"key":"10.1016\/j.ydbio.2019.05.008_bib73","first-page":"263","article-title":"Characterization of the Crumbs homolog 2 (CRB2) gene and analysis of its role in retinitis pigmentosa and Leber congenital amaurosis","volume":"11","author":"van den Hurk","year":"2005","journal-title":"Mol. Vis."},{"key":"10.1016\/j.ydbio.2019.05.008_bib74","doi-asserted-by":"crossref","first-page":"2659","DOI":"10.1093\/hmg\/ddl194","article-title":"Pals1\/Mpp5 is required for correct localization of Crb1 at the subapical region in polarized Muller glia cells","volume":"15","author":"van Rossum","year":"2006","journal-title":"Hum. Mol. Genet."},{"key":"10.1016\/j.ydbio.2019.05.008_bib75","doi-asserted-by":"crossref","first-page":"157","DOI":"10.1016\/j.preteyeres.2018.03.005","article-title":"Non-syndromic retinitis pigmentosa","volume":"66","author":"Verbakel","year":"2018","journal-title":"Prog. Retin. Eye Res."},{"key":"10.1016\/j.ydbio.2019.05.008_bib76","doi-asserted-by":"crossref","first-page":"1448","DOI":"10.1038\/sj.ki.5002579","article-title":"Apical junctional complexes and cell polarity","volume":"72","author":"Wang","year":"2007","journal-title":"Kidney Int."},{"key":"10.1016\/j.ydbio.2019.05.008_bib77","doi-asserted-by":"crossref","first-page":"263","DOI":"10.1016\/j.bbrc.2003.11.122","article-title":"Expression of a novel secretory form (Crb1s) of mouse Crumbs homologue Crb1 in skin development","volume":"313","author":"Watanabe","year":"2004","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"10.1016\/j.ydbio.2019.05.008_bib78","doi-asserted-by":"crossref","first-page":"859","DOI":"10.1007\/s00417-010-1584-6","article-title":"Caspase-3-independent photoreceptor degeneration by N-methyl-N-nitrosourea (MNU) induces morphological and functional changes in the mouse retina","volume":"249","author":"Zulliger","year":"2011","journal-title":"Graefe's archive for clinical and experimental ophthalmology = Albrecht von Graefes Archiv fur klinische und experimentelle Ophthalmologie"}],"container-title":["Developmental Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0012160619300272?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0012160619300272?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,11,2]],"date-time":"2025-11-02T22:49:12Z","timestamp":1762123752000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0012160619300272"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,9]]},"references-count":78,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2019,9]]}},"alternative-id":["S0012160619300272"],"URL":"https:\/\/doi.org\/10.1016\/j.ydbio.2019.05.008","relation":{},"ISSN":["0012-1606"],"issn-type":[{"value":"0012-1606","type":"print"}],"subject":[],"published":{"date-parts":[[2019,9]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Targeted deletion of Crb1\/Crb2 in the optic vesicle models key features of leber congenital amaurosis 8","name":"articletitle","label":"Article Title"},{"value":"Developmental Biology","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.ydbio.2019.05.008","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2019 Elsevier Inc.","name":"copyright","label":"Copyright"}]}}