{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,27]],"date-time":"2026-09-27T22:04:39Z","timestamp":1790546679050,"version":"4.1.0"},"reference-count":62,"publisher":"Oxford University Press (OUP)","issue":"24","license":[{"start":{"date-parts":[[2022,7,21]],"date-time":"2022-07-21T00:00:00Z","timestamp":1658361600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"publisher","award":["431984000"],"award-info":[{"award-number":["431984000"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001659","name":"Deutsche Forschungsgemeinschaft","doi-asserted-by":"publisher","award":["390939984"],"award-info":[{"award-number":["390939984"]}],"id":[{"id":"10.13039\/501100001659","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,12,16]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>The zebrafish pronephros model, using morpholino oligonucleotides (MO) to deplete target genes, has been extensively used to characterize human ciliopathy phenotypes. Recently, discrepancies between MO and genetically defined mutants have questioned this approach. We analyzed zebrafish with mutations in the nphp1-4-8 module to determine the validity of MO-based results. While MO-mediated depletion resulted in glomerular cyst and cloaca malformation, these ciliopathy-typical manifestations were observed at a much lower frequency in zebrafish embryos with defined nphp mutations. All nphp1-4-8 mutant zebrafish were viable and displayed decreased manifestations in the next (F2) generation, lacking maternal RNA contribution. While genetic compensation was further supported by the observation that nphp4-deficient mutants became partially refractory to MO-based nphp4 depletion, zebrafish embryos, lacking one nphp gene, became more sensitive to MO-based depletion of additional nphp genes. Transcriptome analysis of nphp8 mutant embryos revealed an upregulation of the circadian clock genes cry1a and cry5. MO-mediated depletion of cry1a and cry5 caused ciliopathy phenotypes in wild-type embryos, while cry1a and cry5 depletion in maternal zygotic nphp8 mutant embryos increased the frequency of glomerular cysts compared to controls. Importantly, cry1a and cry5 rescued the nephropathy-related phenotypes in nphp1, nphp4 or nphp8-depleted zebrafish embryos. Our results reveal that nphp mutant zebrafish resemble the MO-based phenotypes, albeit at a much lower frequency. Rapid adaption through upregulation of circadian clock genes seems to ameliorate the loss of nphp genes, contributing to phenotypic differences.<\/jats:p>","DOI":"10.1093\/hmg\/ddac160","type":"journal-article","created":{"date-parts":[[2022,7,21]],"date-time":"2022-07-21T14:09:14Z","timestamp":1658412554000},"page":"4143-4158","source":"Crossref","is-referenced-by-count":7,"title":["Clock genes rescue <i>nphp<\/i> mutations in zebrafish"],"prefix":"10.1093","volume":"31","author":[{"given":"Nicolas","family":"Kayser","sequence":"first","affiliation":[{"name":"Renal Division, University Freiburg Medical Center, Faculty of Medicine , Hugstetter Str. 55, Freiburg 79106 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Friedemann","family":"Zaiser","sequence":"additional","affiliation":[{"name":"Renal Division, University Freiburg Medical Center, Faculty of Medicine , Hugstetter Str. 55, Freiburg 79106 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna C","family":"Veenstra","sequence":"additional","affiliation":[{"name":"Renal Division, University Freiburg Medical Center, Faculty of Medicine , Hugstetter Str. 55, Freiburg 79106 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hui","family":"Wang","sequence":"additional","affiliation":[{"name":"Renal Division, University Freiburg Medical Center, Faculty of Medicine , Hugstetter Str. 55, Freiburg 79106 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Burulca","family":"G\u00f6cmen","sequence":"additional","affiliation":[{"name":"Institute of Genetic Epidemiology, Faculty of Medicine and Medical Center, University of Freiburg , Freiburg 79106 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Priska","family":"Eckert","sequence":"additional","affiliation":[{"name":"Renal Division, University Freiburg Medical Center, Faculty of Medicine , Hugstetter Str. 55, Freiburg 79106 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Henriette","family":"Franz","sequence":"additional","affiliation":[{"name":"Department of Biomedicine, University of Basel , Pestalozzistr. 20, Basel CH-4056 , Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"K\u00f6ttgen","sequence":"additional","affiliation":[{"name":"Institute of Genetic Epidemiology, Faculty of Medicine and Medical Center, University of Freiburg , Freiburg 79106 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gerd","family":"Walz","sequence":"additional","affiliation":[{"name":"Renal Division, University Freiburg Medical Center, Faculty of Medicine , Hugstetter Str. 55, Freiburg 79106 , Germany"},{"name":"Signalling Research Centres BIOSS and CIBSS, University of Freiburg , Albertstrasse 19, Freiburg 79104 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Toma A","family":"Yakulov","sequence":"additional","affiliation":[{"name":"Renal Division, University Freiburg Medical Center, Faculty of Medicine , Hugstetter Str. 55, Freiburg 79106 , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2022,7,21]]},"reference":[{"key":"2022121720480476900_ref1","doi-asserted-by":"crossref","first-page":"513","DOI":"10.1016\/j.cell.2011.04.019","article-title":"Mapping the NPHP-JBTS-MKS protein network reveals ciliopathy disease genes and pathways","volume":"145","author":"Sang","year":"2011","journal-title":"Cell"},{"key":"2022121720480476900_ref2","doi-asserted-by":"crossref","first-page":"270","DOI":"10.1681\/ASN.V112270","article-title":"Nephrocystin: gene expression and sequence conservation between human, mouse, and Caenorhabditis elegans","volume":"11","author":"Otto","year":"2000","journal-title":"JASN"},{"key":"2022121720480476900_ref3","doi-asserted-by":"crossref","first-page":"676","DOI":"10.1681\/ASN.2003121025","article-title":"Expression and phenotype analysis of the nephrocystin-1 and nephrocystin-4 homologs in Caenorhabditis elegans","volume":"16","author":"Wolf","year":"2005","journal-title":"JASN"},{"key":"2022121720480476900_ref4","doi-asserted-by":"crossref","first-page":"333","DOI":"10.1016\/j.yexcr.2005.01.008","article-title":"Functional characterization of the C. elegans nephrocystins NPHP-1 and NPHP-4 and their role in cilia and male sensory behaviors","volume":"305","author":"Jauregui","year":"2005","journal-title":"Exp. Cell Res."},{"key":"2022121720480476900_ref5","doi-asserted-by":"crossref","first-page":"782","DOI":"10.1681\/ASN.2009060597","article-title":"Normal ciliogenesis requires synergy between the cystic kidney disease genes MKS-3 and NPHP-4","volume":"21","author":"Williams","year":"2010","journal-title":"JASN"},{"key":"2022121720480476900_ref6","doi-asserted-by":"crossref","first-page":"2942","DOI":"10.1093\/hmg\/ddr198","article-title":"Assessing the pathogenic potential of human nephronophthisis disease-associated NPHP-4 missense mutations in C. elegans","volume":"20","author":"Masyukova","year":"2011","journal-title":"Hum. Mol. Genet."},{"key":"2022121720480476900_ref7","doi-asserted-by":"crossref","first-page":"1023","DOI":"10.1083\/jcb.201012116","article-title":"MKS and NPHP modules cooperate to establish basal body\/transition zone membrane associations and ciliary gate function during ciliogenesis","volume":"192","author":"Williams","year":"2011","journal-title":"J. Cell Biol."},{"key":"2022121720480476900_ref8","first-page":"2592","article-title":"Ciliogenesis in Caenorhabditis elegans requires genetic interactions between ciliary middle segment localized NPHP-2 (inversin) and transition zone-associated proteins","volume":"125","author":"Warburton-Pitt","year":"2012","journal-title":"J. Cell Sci."},{"key":"2022121720480476900_ref9","doi-asserted-by":"crossref","first-page":"149","DOI":"10.1038\/ng1097-149","article-title":"A novel gene encoding an SH3 domain protein is mutated in nephronophthisis type 1","volume":"17","author":"Hildebrandt","year":"1997","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref10","doi-asserted-by":"crossref","first-page":"3368","DOI":"10.1093\/hmg\/ddn231","article-title":"Targeted disruption of Nphp1 causes male infertility due to defects in the later steps of sperm morphogenesis in mice","volume":"17","author":"Jiang","year":"2008","journal-title":"Hum. Mol. Genet."},{"key":"2022121720480476900_ref11","doi-asserted-by":"crossref","first-page":"482","DOI":"10.1093\/hmg\/ddq494","article-title":"NPHP4 is necessary for normal photoreceptor ribbon synapse maintenance and outer segment formation, and for sperm development","volume":"20","author":"Won","year":"2011","journal-title":"Hum. Mol. Genet."},{"key":"2022121720480476900_ref12","doi-asserted-by":"crossref","first-page":"882","DOI":"10.1038\/ng2069","article-title":"Mutations in the gene encoding the basal body protein RPGRIP1L, a nephrocystin-4 interactor, cause Joubert syndrome","volume":"39","author":"Arts","year":"2007","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref13","doi-asserted-by":"crossref","first-page":"875","DOI":"10.1038\/ng2039","article-title":"The ciliary gene RPGRIP1L is mutated in cerebello-oculo-renal syndrome (Joubert syndrome type B) and Meckel syndrome","volume":"39","author":"Delous","year":"2007","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref14","doi-asserted-by":"crossref","first-page":"2569","DOI":"10.1242\/dev.003715","article-title":"Ftm is a novel basal body protein of cilia involved in Shh signalling","volume":"134","author":"Vierkotten","year":"2007","journal-title":"Development"},{"key":"2022121720480476900_ref15","doi-asserted-by":"crossref","first-page":"3602","DOI":"10.1096\/fj.12-203554","article-title":"Advances in understanding the peripheral circadian clocks","volume":"26","author":"Richards","year":"2012","journal-title":"FASEB J."},{"key":"2022121720480476900_ref16","doi-asserted-by":"crossref","first-page":"42","DOI":"10.1007\/s11892-019-1156-z","article-title":"Circadian clock genes in diabetic kidney disease (DKD)","volume":"19","author":"Olaoye","year":"2019","journal-title":"Curr. Diab. Rep."},{"key":"2022121720480476900_ref17","doi-asserted-by":"crossref","first-page":"90","DOI":"10.1016\/j.tcb.2013.07.002","article-title":"Molecular architecture of the mammalian circadian clock","volume":"24","author":"Partch","year":"2014","journal-title":"Trends Cell Biol."},{"key":"2022121720480476900_ref18","doi-asserted-by":"crossref","first-page":"D1009","DOI":"10.1093\/nar\/gks1161","article-title":"CircaDB: a database of mammalian circadian gene expression profiles","volume":"41","author":"Pizarro","year":"2013","journal-title":"Nucleic Acids Res."},{"key":"2022121720480476900_ref19","first-page":"167","article-title":"Effects of 5\/6 nephrectomy on renal function and blood pressure in mice","volume":"4","author":"Gava","year":"2012","journal-title":"Int. J. Physiol. Pathophysiol. Pharmacol."},{"key":"2022121720480476900_ref20","doi-asserted-by":"crossref","first-page":"98","DOI":"10.1097\/00005344-200307000-00015","article-title":"Influence of two doses of irbesartan on non-dipper circadian blood pressure rhythm in salt-sensitive black hypertensives under high salt diet","volume":"42","author":"Pol\u00f3nia","year":"2003","journal-title":"J. Cardiovasc. Pharmacol."},{"key":"2022121720480476900_ref21","doi-asserted-by":"crossref","first-page":"555","DOI":"10.1111\/nep.12111","article-title":"Altered diurnal variation and localization of clock proteins in the remnant kidney of 5\/6 nephrectomy rats","volume":"18","author":"Huang","year":"2013","journal-title":"Nephrology (Carlton)"},{"key":"2022121720480476900_ref22","doi-asserted-by":"crossref","first-page":"1430","DOI":"10.1681\/ASN.2013060641","article-title":"Local renal circadian clocks control fluid-electrolyte homeostasis and BP","volume":"25","author":"Tokonami","year":"2014","journal-title":"J. Am. Soc. Nephrol."},{"key":"2022121720480476900_ref23","doi-asserted-by":"crossref","first-page":"230","DOI":"10.1038\/nature14580","article-title":"Genetic compensation induced by deleterious mutations but not gene knockdowns","volume":"524","author":"Rossi","year":"2015","journal-title":"Nature"},{"key":"2022121720480476900_ref24","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.1006780","article-title":"Genetic compensation: a phenomenon in search of mechanisms","volume":"13","author":"El-Brolosy","year":"2017","journal-title":"PLoS Genet."},{"key":"2022121720480476900_ref25","doi-asserted-by":"crossref","first-page":"193","DOI":"10.1038\/s41586-019-1064-z","article-title":"Genetic compensation triggered by mutant mRNA degradation","volume":"568","author":"El-Brolosy","year":"2019","journal-title":"Nature"},{"key":"2022121720480476900_ref26","doi-asserted-by":"crossref","first-page":"550","DOI":"10.1186\/s13059-014-0550-8","article-title":"Moderated estimation of fold change and dispersion for RNA-seq data with DESeq2","volume":"15","author":"Love","year":"2014","journal-title":"Genome Biol."},{"key":"2022121720480476900_ref27","doi-asserted-by":"crossref","first-page":"196","DOI":"10.1038\/s41598-018-37879-8","article-title":"The clock components Period2, Cryptochrome1a, and Cryptochrome2a function in establishing light-dependent behavioral rhythms and\/or total activity levels in zebrafish","volume":"9","author":"Hirayama","year":"2019","journal-title":"Sci. Rep."},{"key":"2022121720480476900_ref28","doi-asserted-by":"crossref","first-page":"2794","DOI":"10.4161\/cc.8.17.9447","article-title":"Common light signaling pathways controlling DNA repair and circadian clock entrainment in zebrafish","volume":"8","author":"Hirayama","year":"2009","journal-title":"Cell Cycle"},{"key":"2022121720480476900_ref29","doi-asserted-by":"crossref","first-page":"2611","DOI":"10.1093\/hmg\/ddr164","article-title":"Control of the Wnt pathways by nephrocystin-4 is required for morphogenesis of the zebrafish pronephros","volume":"20","author":"Burckl\u00e9","year":"2011","journal-title":"Hum. Mol. Genet."},{"key":"2022121720480476900_ref30","doi-asserted-by":"crossref","first-page":"3119","DOI":"10.1093\/hmg\/ddr214","article-title":"Nephrocystin-4 is required for pronephric duct-dependent cloaca formation in zebrafish","volume":"20","author":"Slanchev","year":"2011","journal-title":"Hum. Mol. Genet."},{"key":"2022121720480476900_ref31","doi-asserted-by":"crossref","first-page":"1161","DOI":"10.1086\/344395","article-title":"A gene mutated in nephronophthisis and retinitis pigmentosa encodes a novel protein, nephroretinin, conserved in evolution","volume":"71","author":"Otto","year":"2002","journal-title":"Am. J. Hum. Genet."},{"key":"2022121720480476900_ref32","doi-asserted-by":"crossref","first-page":"767","DOI":"10.1016\/j.cmet.2014.04.009","article-title":"Hypomorphism for RPGRIP1L, a ciliary gene vicinal to the FTO locus, causes increased adiposity in mice","volume":"19","author":"Stratigopoulos","year":"2014","journal-title":"Cell Metab."},{"key":"2022121720480476900_ref33","doi-asserted-by":"crossref","first-page":"1897","DOI":"10.1172\/JCI85526","article-title":"Hypomorphism of Fto and Rpgrip1l causes obesity in mice","volume":"126","author":"Stratigopoulos","year":"2016","journal-title":"J. Clin. Invest."},{"key":"2022121720480476900_ref34","doi-asserted-by":"crossref","first-page":"3946","DOI":"10.1096\/fj.201701216R","article-title":"The role of Rpgrip1l, a component of the primary cilium, in adipocyte development and function","volume":"32","author":"Carli","year":"2018","journal-title":"FASEB J."},{"key":"2022121720480476900_ref35","doi-asserted-by":"crossref","first-page":"123337","DOI":"10.1172\/jci.insight.123337","article-title":"Ciliary gene RPGRIP1L is required for hypothalamic arcuate neuron development","volume":"4","author":"Wang","year":"2019","journal-title":"JCI Insight"},{"key":"2022121720480476900_ref36","doi-asserted-by":"crossref","first-page":"67","DOI":"10.1038\/nm.2061","article-title":"Salt-sensitive hypertension in circadian clock-deficient Cry-null mice involves dysregulated adrenal Hsd3b6","volume":"16","author":"Doi","year":"2010","journal-title":"Nat. Med."},{"key":"2022121720480476900_ref37","doi-asserted-by":"crossref","first-page":"2423","DOI":"10.1172\/JCI36908","article-title":"The circadian clock protein Period 1 regulates expression of the renal epithelial sodium channel in mice","volume":"119","author":"Gumz","year":"2009","journal-title":"J. Clin. Invest."},{"key":"2022121720480476900_ref38","doi-asserted-by":"crossref","first-page":"253","DOI":"10.3389\/fphys.2013.00253","article-title":"Role of Per1 and the mineralocorticoid receptor in the coordinate regulation of \u03b1ENaC in renal cortical collecting duct cells","volume":"4","author":"Richards","year":"2013","journal-title":"Front. Physiol."},{"key":"2022121720480476900_ref39","doi-asserted-by":"crossref","first-page":"11791","DOI":"10.1074\/jbc.M113.531095","article-title":"A role for the circadian clock protein Per1 in the regulation of the NaCl co-transporter (NCC) and the with-no-lysine kinase (WNK) cascade in mouse distal convoluted tubule cells","volume":"289","author":"Richards","year":"2014","journal-title":"J. Biol. Chem."},{"key":"2022121720480476900_ref40","doi-asserted-by":"crossref","first-page":"255","DOI":"10.1016\/j.lfs.2014.03.028","article-title":"Tissue-specific and time-dependent regulation of the endothelin axis by the circadian clock protein Per1","volume":"118","author":"Richards","year":"2014","journal-title":"Life Sci."},{"key":"2022121720480476900_ref41","doi-asserted-by":"crossref","first-page":"622","DOI":"10.1016\/j.bbagrm.2010.09.003","article-title":"Regulation of \u03b1ENaC expression by the circadian clock protein Period 1 in mpkCCD(c14) cells","volume":"1799","author":"Gumz","year":"2010","journal-title":"Biochim. Biophys. Acta"},{"key":"2022121720480476900_ref42","doi-asserted-by":"crossref","first-page":"72","DOI":"10.1111\/apha.12804","article-title":"Desoxycorticosterone pivalate-salt treatment leads to non-dipping hypertension in Per1 knockout mice","volume":"220","author":"Solocinski","year":"2017","journal-title":"Acta Physiol (Oxf.)"},{"key":"2022121720480476900_ref43","doi-asserted-by":"crossref","first-page":"455","DOI":"10.1038\/ng1216","article-title":"Mutations in a novel gene, NPHP3, cause adolescent nephronophthisis, tapeto-retinal degeneration and hepatic fibrosis","volume":"34","author":"Olbrich","year":"2003","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref44","doi-asserted-by":"crossref","first-page":"413","DOI":"10.1038\/ng1217","article-title":"Mutations in INVS encoding inversin cause nephronophthisis type 2, linking renal cystic disease to the function of primary cilia and left-right axis determination","volume":"34","author":"Otto","year":"2003","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref45","doi-asserted-by":"crossref","first-page":"674","DOI":"10.1038\/ng1786","article-title":"The centrosomal protein nephrocystin-6 is mutated in Joubert syndrome and activates transcription factor ATF4","volume":"38","author":"Sayer","year":"2006","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref46","doi-asserted-by":"crossref","first-page":"619","DOI":"10.1038\/ng.594","article-title":"Mutations in TMEM216 perturb ciliogenesis and cause Joubert, Meckel and related syndromes","volume":"42","author":"Valente","year":"2010","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref47","doi-asserted-by":"crossref","first-page":"533","DOI":"10.1016\/j.cell.2012.06.028","article-title":"Exome capture reveals ZNF423 and CEP164 mutations, linking renal ciliopathies to DNA damage response signaling","volume":"150","author":"Chaki","year":"2012","journal-title":"Cell"},{"key":"2022121720480476900_ref48","doi-asserted-by":"crossref","first-page":"951","DOI":"10.1038\/ng.2681","article-title":"ANKS6 is a central component of a nephronophthisis module linking NEK8 to INVS and NPHP3","volume":"45","author":"Hoff","year":"2013","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref49","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.1004594","article-title":"Nephronophthisis-associated CEP164 regulates cell cycle progression, apoptosis and epithelial-to-mesenchymal transition","volume":"10","author":"Slaats","year":"2014","journal-title":"PLoS Genet."},{"key":"2022121720480476900_ref50","doi-asserted-by":"crossref","first-page":"45","DOI":"10.1016\/j.ajhg.2018.11.003","article-title":"Mutations of ADAMTS9 cause nephronophthisis-related ciliopathy","volume":"104","author":"Choi","year":"2019","journal-title":"Am. J. Hum. Genet."},{"key":"2022121720480476900_ref51","doi-asserted-by":"crossref","first-page":"1133","DOI":"10.12688\/f1000research.15511.1","article-title":"Using zebrafish to study the function of nephronophthisis and related ciliopathy genes","volume":"7","author":"Molinari","year":"2018","journal-title":"F1000Res"},{"key":"2022121720480476900_ref52","volume-title":"The Zebrafish Book: A Guide for the Laboratory Use of Zebrafish","author":"Westerfield","year":"1995","edition":"3rd"},{"key":"2022121720480476900_ref53","doi-asserted-by":"crossref","first-page":"3660","DOI":"10.1038\/s41467-018-06094-4","article-title":"CXCL12 and MYC control energy metabolism to support adaptive responses after kidney injury","volume":"9","author":"Yakulov","year":"2018","journal-title":"Nat. Commun."},{"key":"2022121720480476900_ref54","doi-asserted-by":"crossref","first-page":"W272","DOI":"10.1093\/nar\/gkw398","article-title":"CHOPCHOP v2: a web tool for the next generation of CRISPR genome engineering","volume":"44","author":"Labun","year":"2016","journal-title":"Nucleic Acids Res."},{"key":"2022121720480476900_ref55","doi-asserted-by":"crossref","first-page":"355","DOI":"10.1016\/B978-0-12-801185-0.00017-9","article-title":"Cas9-based genome editing in Xenopus tropicalis","volume":"546","author":"Nakayama","year":"2014","journal-title":"Methods Enzymol."},{"key":"2022121720480476900_ref56","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pgen.0030078","article-title":"p53 Activation by knockdown technologies","volume":"3","author":"Robu","year":"2007","journal-title":"PLoS Genet."},{"key":"2022121720480476900_ref57","doi-asserted-by":"crossref","first-page":"739","DOI":"10.1038\/ng.366","article-title":"A common allele in RPGRIP1L is a modifier of retinal degeneration in ciliopathies","volume":"41","author":"Khanna","year":"2009","journal-title":"Nat. Genet."},{"key":"2022121720480476900_ref58","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":"Schindelin","year":"2012","journal-title":"Nat. Methods"},{"key":"2022121720480476900_ref59","doi-asserted-by":"crossref","first-page":"F826","DOI":"10.1152\/ajprenal.00610.2020","article-title":"Single-cell mRNA profiling reveals changes in solute carrier expression and suggests a metabolic switch during zebrafish pronephros development","volume":"320","author":"Schoels","year":"2021","journal-title":"Am. J. Physiol. Renal Physiol."},{"key":"2022121720480476900_ref60","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1093\/bioinformatics\/bts635","article-title":"STAR: ultrafast universal RNA-seq aligner","volume":"29","author":"Dobin","year":"2013","journal-title":"Bioinformatics"},{"key":"2022121720480476900_ref61","doi-asserted-by":"crossref","first-page":"323","DOI":"10.1186\/1471-2105-12-323","article-title":"RSEM: accurate transcript quantification from RNA-Seq data with or without a reference genome","volume":"12","author":"Li","year":"2011","journal-title":"BMC Bioinformatics"},{"key":"2022121720480476900_ref62","doi-asserted-by":"crossref","first-page":"e31","DOI":"10.1158\/0008-5472.CAN-17-0337","article-title":"Variant review with the integrative genomics viewer","volume":"77","author":"Robinson","year":"2017","journal-title":"Cancer Res."}],"container-title":["Human Molecular Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/hmg\/advance-article-pdf\/doi\/10.1093\/hmg\/ddac160\/45197849\/ddac160.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/hmg\/article-pdf\/31\/24\/4143\/48000054\/ddac160.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/hmg\/article-pdf\/31\/24\/4143\/48000054\/ddac160.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,12,17]],"date-time":"2022-12-17T20:48:37Z","timestamp":1671310117000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/hmg\/article\/31\/24\/4143\/6647922"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,7,21]]},"references-count":62,"journal-issue":{"issue":"24","published-online":{"date-parts":[[2022,7,21]]},"published-print":{"date-parts":[[2022,12,16]]}},"URL":"https:\/\/doi.org\/10.1093\/hmg\/ddac160","relation":{},"ISSN":["0964-6906","1460-2083"],"issn-type":[{"value":"0964-6906","type":"print"},{"value":"1460-2083","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2022,12,15]]},"published":{"date-parts":[[2022,7,21]]}}}