{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,22]],"date-time":"2026-10-22T06:07:03Z","timestamp":1792649223908,"version":"4.5.0"},"reference-count":46,"publisher":"Elsevier","isbn-type":[{"value":"9780443185885","type":"print"}],"license":[{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-017"},{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-037"},{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-012"},{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-029"},{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"stm-asf","delay-in-days":0,"URL":"https:\/\/doi.org\/10.15223\/policy-004"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023]]},"DOI":"10.1016\/bs.mcb.2023.02.016","type":"book-chapter","created":{"date-parts":[[2023,3,27]],"date-time":"2023-03-27T15:41:40Z","timestamp":1679931700000},"page":"139-158","source":"Crossref","is-referenced-by-count":4,"title":["Modeling ciliopathies in patient-derived primary cells"],"prefix":"10.1016","member":"78","reference":[{"issue":"735","key":"10.1016\/bs.mcb.2023.02.016_bb0010","first-page":"1","article-title":"Activation of canonical Wnt signalling is required for TGF-\u03b2-mediated fibrosis","volume":"3","author":"Akhmetshina","year":"2012","journal-title":"Nature Communications"},{"issue":"1","key":"10.1016\/bs.mcb.2023.02.016_bb0015","doi-asserted-by":"crossref","first-page":"20","DOI":"10.1007\/s12035-012-8245-1","article-title":"Primary skin fibroblasts as a model of Parkinson's disease","volume":"46","author":"Auburger","year":"2012","journal-title":"Molecular Neurobiology"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0020","article-title":"Braun and Friedhelm Hildebrandt","volume":"9","author":"Braun","year":"2017","journal-title":"Cold Spring Harbor Perspective in Biology"},{"issue":"10","key":"10.1016\/bs.mcb.2023.02.016_bb0025","doi-asserted-by":"crossref","first-page":"1080","DOI":"10.4252\/wjsc.v12.i10.1080","article-title":"Urine-derived stem\/progenitor cells: A focus on their characterization and potential","volume":"12","author":"Burdeyron","year":"2020","journal-title":"World Journal of Stem Cells"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0030","first-page":"1","article-title":"Generation of human kidney tubuloids from tissue and urine","volume":"170","author":"Calandrini","year":"2021","journal-title":"Journal of Visualized Experiments"},{"issue":"6","key":"10.1016\/bs.mcb.2023.02.016_bb0035","doi-asserted-by":"crossref","first-page":"1756","DOI":"10.1038\/s41596-019-0160-8","article-title":"High-resolution 3D imaging of fixed and cleared organoids","volume":"14","author":"Dekkers","year":"2019","journal-title":"Nature Protocols"},{"issue":"2","key":"10.1016\/bs.mcb.2023.02.016_bb0040","doi-asserted-by":"crossref","first-page":"383","DOI":"10.1038\/ki.1984.28","article-title":"Tissue culture of human kidney epithelial cells of proximal tubule origin","volume":"25","author":"Detrisac","year":"1984","journal-title":"Kidney International"},{"issue":"10","key":"10.1016\/bs.mcb.2023.02.016_bb0045","doi-asserted-by":"crossref","first-page":"618","DOI":"10.1007\/s002040000173","article-title":"Cultures of exfoliated epithelial cells from different locations of the human urinary tract and the renal tubular system","volume":"74","author":"D\u00f6rrenhaus","year":"2000","journal-title":"Archives of Toxicology"},{"issue":"11","key":"10.1016\/bs.mcb.2023.02.016_bb0050","doi-asserted-by":"crossref","first-page":"880","DOI":"10.1038\/nrm2278","article-title":"When cilia go bad: Cilia defects and ciliopathies","volume":"8","author":"Fliegauf","year":"2007","journal-title":"Nature Reviews Molecular Cell Biology"},{"issue":"1","key":"10.1016\/bs.mcb.2023.02.016_bb0055","doi-asserted-by":"crossref","first-page":"62","DOI":"10.4161\/org.28910","article-title":"The primary cilium: Guardian of organ development and homeostasis","volume":"10","author":"Fry","year":"2014","journal-title":"Organogenesis"},{"issue":"4","key":"10.1016\/bs.mcb.2023.02.016_bb0060","doi-asserted-by":"crossref","first-page":"2023","DOI":"10.1038\/s41596-020-00479-w","article-title":"Culture and analysis of kidney tubuloids and perfused tubuloid cells-on-a-chip","volume":"16","author":"Gijzen","year":"2021","journal-title":"Nature Protocols"},{"issue":"3","key":"10.1016\/bs.mcb.2023.02.016_bb0065","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1371\/journal.pgen.1005894","article-title":"Novel NEK8 mutations cause severe syndromic renal cystic dysplasia through YAP dysregulation","volume":"12","author":"Grampa","year":"2016","journal-title":"PLoS Genetics"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0070","doi-asserted-by":"crossref","first-page":"153","DOI":"10.3181\/00379727-161-40509","article-title":"Short-term culture of epithelial cells from urine of adults","volume":"3","author":"Herz","year":"1979","journal-title":"Proceedings of the Society for Experimental Biology and Medicine"},{"issue":"9722","key":"10.1016\/bs.mcb.2023.02.016_bb0075","doi-asserted-by":"crossref","first-page":"1287","DOI":"10.1016\/S0140-6736(10)60236-X","article-title":"Genetic kidney diseases","volume":"375","author":"Hildebrandt","year":"2010","journal-title":"The Lancet"},{"issue":"16","key":"10.1016\/bs.mcb.2023.02.016_bb0080","doi-asserted-by":"crossref","first-page":"1533","DOI":"10.1056\/NEJMra1010172","article-title":"Ciliopathies","volume":"364","author":"Hildebrandt","year":"2011","journal-title":"The New England Journal of Medicine"},{"issue":"8","key":"10.1016\/bs.mcb.2023.02.016_bb0085","doi-asserted-by":"crossref","first-page":"2724","DOI":"10.1002\/art.34444","article-title":"Hedgehog signaling controls fibroblast activation and tissue fibrosis in systemic sclerosis","volume":"64","author":"Horn","year":"2012","journal-title":"Arthritis and Rheumatism"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0090","doi-asserted-by":"crossref","first-page":"5","DOI":"10.1186\/s13630-017-0045-9","article-title":"Fixation methods can differentially affect ciliary protein immunolabeling","volume":"6","author":"Hua","year":"2017","journal-title":"Cilia"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0095","doi-asserted-by":"crossref","first-page":"225","DOI":"10.1111\/j.1749-6632.1994.tb44412.x","article-title":"Use of cultured fibroblasts in elucidating the pathophysiology and diagnosis of Alzheimer's disease","volume":"747","author":"Huang","year":"1994","journal-title":"Annals of the New York Academy of Sciences"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0100","series-title":"In vitro cell developmental biology\u2014Animal","first-page":"364","article-title":"Reconstruction of tubular structures in three-dimensional collagen gel culture using proximal tubular epithelial cells voided in human urine","volume":"Vol. 39","author":"Inoue","year":"2003"},{"issue":"7","key":"10.1016\/bs.mcb.2023.02.016_bb0105","doi-asserted-by":"crossref","first-page":"1","DOI":"10.15252\/emmm.202013067","article-title":"Cysteamine\u2013bicalutamide combination therapy corrects proximal tubule phenotype in cystinosis","volume":"13","author":"Jamalpoor","year":"2021","journal-title":"EMBO Molecular Medicine"},{"issue":"8","key":"10.1016\/bs.mcb.2023.02.016_bb0110","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0043584","article-title":"Distinct mesenchymal alterations in N-cadherin and E-cadherin positive primary renal epithelial cells","volume":"7","author":"Keller","year":"2012","journal-title":"PLoS One"},{"issue":"6","key":"10.1016\/bs.mcb.2023.02.016_bb0115","doi-asserted-by":"crossref","first-page":"1079","DOI":"10.1089\/scd.2007.0247","article-title":"The ROCK inhibitor Y-27632 enhances the survival rate of human embryonic stem cells following cryopreservation","volume":"17","author":"Li","year":"2008","journal-title":"Stem Cells and Development"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0120","doi-asserted-by":"crossref","first-page":"a028209","DOI":"10.1101\/cshperspect.a028209","article-title":"Ciliary mechanisms of cyst formation in polycystic kidney disease","volume":"9","author":"Ma","year":"2017","journal-title":"Cold Spring Harbor Perspectives in Biology"},{"issue":"3","key":"10.1016\/bs.mcb.2023.02.016_bb0125","doi-asserted-by":"crossref","first-page":"410","DOI":"10.1053\/j.ajkd.2020.08.012","article-title":"Ciliopathies and the kidney: A review","volume":"77","author":"McConnachie","year":"2021","journal-title":"American Journal of Kidney Diseases"},{"issue":"5","key":"10.1016\/bs.mcb.2023.02.016_bb0130","doi-asserted-by":"crossref","first-page":"555","DOI":"10.1002\/ana.21492","article-title":"Mitochondrial function and morphology are impaired in parkin-mutant fibroblasts","volume":"64","author":"Mortiboys","year":"2008","journal-title":"Annals of Neurology"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0135","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1155\/2015\/362562","article-title":"Human urine as a noninvasive source of kidney cells","volume":"362562","author":"Oliveira Arcolino","year":"2015","journal-title":"Stem Cells International"},{"issue":"1","key":"10.1016\/bs.mcb.2023.02.016_bb0140","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1016\/S0955-0674(02)00012-1","article-title":"The vertebrate primary cilium is a sensory organelle","volume":"15","author":"Pazour","year":"2003","journal-title":"Current Opinion in Cell Biology"},{"issue":"9","key":"10.1016\/bs.mcb.2023.02.016_bb0145","doi-asserted-by":"crossref","first-page":"533","DOI":"10.1038\/nrm.2017.60","article-title":"Genes and molecular pathways underpinning ciliopathies","volume":"18","author":"Reiter","year":"2017","journal-title":"Nature Reviews Molecular Cell Biology"},{"issue":"7","key":"10.1016\/bs.mcb.2023.02.016_bb0150","doi-asserted-by":"crossref","first-page":"711","DOI":"10.1038\/ncb3370","article-title":"Cilium assembly and disassembly","volume":"18","author":"S\u00e1nchez","year":"2016","journal-title":"Nature Cell Biology"},{"issue":"7244","key":"10.1016\/bs.mcb.2023.02.016_bb0155","doi-asserted-by":"crossref","first-page":"262","DOI":"10.1038\/nature07935","article-title":"Single Lgr5 stem cells build crypt-villus structures in vitro without a mesenchymal niche","volume":"459","author":"Sato","year":"2009","journal-title":"Nature"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0160","doi-asserted-by":"crossref","first-page":"211","DOI":"10.1146\/annurev-pathmechdis-012419-032611","article-title":"Human organoids: Tools for understanding biology and treating diseases","volume":"15","author":"Schutgens","year":"2020","journal-title":"Annual Review of Pathology: Mechanisms of Disease"},{"issue":"3","key":"10.1016\/bs.mcb.2023.02.016_bb0165","doi-asserted-by":"crossref","first-page":"303","DOI":"10.1038\/s41587-019-0048-8","article-title":"Tubuloids derived from human adult kidney and urine for personalized disease modeling","volume":"37","author":"Schutgens","year":"2019","journal-title":"Nature Biotechnology"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0170","first-page":"1","article-title":"Biallelic ANKS6 mutations cause late-onset ciliopathy with chronic kidney disease through YAP dysregulation","author":"Schwarz","year":"2021","journal-title":"Human Molecular Genetics"},{"issue":"2","key":"10.1016\/bs.mcb.2023.02.016_bb0175","doi-asserted-by":"crossref","first-page":"359","DOI":"10.1042\/BCJ20190513","article-title":"Proteostasis regulators modulate proteasomal activity and gene expression to attenuate multiple phenotypes in Fabry disease","volume":"477","author":"Seemann","year":"2020","journal-title":"Biochemical Journal"},{"issue":"2","key":"10.1016\/bs.mcb.2023.02.016_bb0180","doi-asserted-by":"crossref","first-page":"384","DOI":"10.1016\/j.celrep.2017.06.045","article-title":"In vitro modeling using ciliopathy-patient-derived cells reveals distinct cilia dysfunctions caused by CEP290 mutations","volume":"20","author":"Shimada","year":"2017","journal-title":"Cell Reports"},{"issue":"23","key":"10.1016\/bs.mcb.2023.02.016_bb0185","doi-asserted-by":"crossref","first-page":"4657","DOI":"10.1093\/hmg\/ddx347","article-title":"A human patient-derived cellular model of Joubert syndrome reveals ciliary defects which can be rescued with targeted therapies","volume":"26","author":"Srivastava","year":"2017","journal-title":"Human Molecular Genetics"},{"issue":"6","key":"10.1016\/bs.mcb.2023.02.016_bb0190","doi-asserted-by":"crossref","first-page":"1067","DOI":"10.1016\/j.jprot.2009.10.012","article-title":"A single fixation protocol for proteome-wide immunofluorescence localization studies","volume":"73","author":"Stadler","year":"2010","journal-title":"Journal of Proteomics"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0195","doi-asserted-by":"crossref","DOI":"10.3389\/fcell.2021.653138","article-title":"Renal ciliopathies: Sorting out therapeutic approaches for nephronophthisis","volume":"9","author":"Stokman","year":"2021","journal-title":"Frontiers in Cell and Developmental Biology"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0200","doi-asserted-by":"crossref","first-page":"231","DOI":"10.1038\/239231a0","article-title":"Culture of cells from the urine of newborn children","volume":"239","author":"Sutherland","year":"1972","journal-title":"Nature"},{"issue":"5","key":"10.1016\/bs.mcb.2023.02.016_bb0205","doi-asserted-by":"crossref","first-page":"861","DOI":"10.1016\/j.cell.2007.11.019","article-title":"Induction of pluripotent stem cells from adult human fibroblasts by defined factors","volume":"131","author":"Takahashi","year":"2007","journal-title":"Cell"},{"issue":"1","key":"10.1016\/bs.mcb.2023.02.016_bb0210","doi-asserted-by":"crossref","first-page":"51","DOI":"10.1016\/0531-5565(93)90019-A","article-title":"Growth properties of familial alzheimer skin fibroblasts during in vitro aging","volume":"28","author":"Tesco","year":"1993","journal-title":"Experimental Gerontology"},{"issue":"3","key":"10.1016\/bs.mcb.2023.02.016_bb0215","doi-asserted-by":"crossref","first-page":"527","DOI":"10.1016\/0092-8674(79)90261-7","article-title":"Centriole ciliation is related to quiescence and DNA synthesis in 3T3 cells","volume":"17","author":"Tucker","year":"1979","journal-title":"Cell"},{"issue":"2","key":"10.1016\/bs.mcb.2023.02.016_bb0220","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0001565","article-title":"Reproducible, ultra high-throughput formation of multicellular organization from single cell suspension-derived human embryonic stem cell aggregates","volume":"3","author":"Ungrin","year":"2008","journal-title":"PLoS One"},{"key":"10.1016\/bs.mcb.2023.02.016_bb0225","doi-asserted-by":"crossref","first-page":"681","DOI":"10.1038\/nbt1310","article-title":"A ROCK inhibitor permits survival of dissociated human embryonic stem cells","volume":"25","author":"Watanabe","year":"2007","journal-title":"Nature Biotechnology"},{"issue":"12","key":"10.1016\/bs.mcb.2023.02.016_bb0230","doi-asserted-by":"crossref","first-page":"2080","DOI":"10.1038\/nprot.2012.115","article-title":"Generation of human induced pluripotent stem cells from urine samples","volume":"7","author":"Zhou","year":"2012","journal-title":"Nature Protocols"},{"issue":"7","key":"10.1016\/bs.mcb.2023.02.016_bb0235","doi-asserted-by":"crossref","first-page":"1221","DOI":"10.1681\/ASN.2011010106","article-title":"Generation of induced pluripotent stem cells from urine","volume":"22","author":"Zhou","year":"2011","journal-title":"Journal of the American Society of Nephrology"}],"container-title":["Methods in Cell Biology","Cilia: From Mechanisms to Disease - Part B"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0091679X23000602?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S0091679X23000602?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,10,27]],"date-time":"2025-10-27T08:07:30Z","timestamp":1761552450000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S0091679X23000602"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023]]},"ISBN":["9780443185885"],"references-count":46,"URL":"https:\/\/doi.org\/10.1016\/bs.mcb.2023.02.016","relation":{},"ISSN":["0091-679X"],"issn-type":[{"value":"0091-679X","type":"print"}],"subject":[],"published":{"date-parts":[[2023]]}}}