{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T12:23:46Z","timestamp":1787747026754,"version":"build-2784847793"},"reference-count":45,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"8","license":[{"start":{"date-parts":[[2026,2,10]],"date-time":"2026-02-10T00:00:00Z","timestamp":1770681600000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000062","name":"National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"crossref","award":["R01DK115752"],"award-info":[{"award-number":["R01DK115752"]}],"id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"crossref"}]},{"name":"National Institute of Child Health and Human Development","award":["R01HD111399"],"award-info":[{"award-number":["R01HD111399"]}]},{"DOI":"10.13039\/100000738","name":"US Department of Veterans Affairs","doi-asserted-by":"crossref","award":["1-I01-BX006266-01"],"award-info":[{"award-number":["1-I01-BX006266-01"]}],"id":[{"id":"10.13039\/100000738","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":["JASN"],"published-print":{"date-parts":[[2026,8]]},"abstract":"\n Key Points<\/jats:title>\n \n \n \n A HaloTag knock-in resource allows direct visualization of endogenous polycystin-2 (PC2), enabling quantitative analysis of its localization, turnover, and transport dynamics.<\/jats:p>\n <\/jats:list-item>\n \n \n PC2-HaloTag labeling establishes PC1-dependent and Tulp3-dependent control of PC2 ciliary targeting\n in vivo<\/jats:italic>\n and in cells.\n <\/jats:p>\n <\/jats:list-item>\n <\/jats:list>\n <\/jats:p>\n <\/jats:sec>\n \n Background<\/jats:title>\n \n Polycystin-2, encoded by\n PKD2<\/jats:italic>\n , is a cation channel essential for kidney physiology. Dysfunction of Pkd2 causes autosomal dominant polycystic kidney disease. Currently, our understanding of cystogenesis in the kidney is limited by the difficulty of visualizing the localization and molecular functions of endogenous polycystin proteins.\n <\/jats:p>\n <\/jats:sec>\n \n Methods<\/jats:title>\n \n Using clustered regularly interspaced short palindromic repeats\/Cas9, we engineered a\n Pkd2<\/jats:italic>\n HaloTag knock-in mouse (referred to as\n Pkd2<\/jats:italic>\n c-Halo<\/jats:sup>\n ) and derived tsSV40-immortalized\n Pkd2<\/jats:italic>\n c-Halo<\/jats:sup>\n renal epithelial cell lines. We optimized HaloTag labeling for\n in vivo<\/jats:italic>\n and\n in vitro<\/jats:italic>\n applications, demonstrating its use in confocal and live cell microscopy, pulse-chase assays, affinity isolation, and\n in vivo<\/jats:italic>\n imaging of Pkd2 protein localization after kidney injury and in disease-relevant\n Tulp3<\/jats:italic>\n R400W\/R400W<\/jats:sup>\n and\n Pkd1<\/jats:italic>\n null<\/jats:sup>\n mutant backgrounds.\n <\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n \n Homozygous\n Pkd2<\/jats:italic>\n c-Halo<\/jats:sup>\n mice were viable, fertile, and phenotypically normal, confirming that the C-terminal HaloTag did not disrupt Pkd2 function. Pkd2-c-Halo was detected by Western blotting and localized to endoplasmic reticulum and the primary cilium. Labeling occurred within 30 minutes and plateaued by 3 hours at doses of \u22652 nmol\/mouse and \u226525 nM\n in vivo<\/jats:italic>\n and in cultured cells, respectively. Pulse-chase analysis showed complete Pkd2-c-Halo turnover within 48 hours in the cilia of the choroid plexus\n in vivo<\/jats:italic>\n and 24 hours in cultured renal epithelial cells. HaloTrap affinity resin purified the endogenous Pkd2-c-Halo from cells and tissues efficiently for protein complex analysis. While kidney injury is known to accelerate cyst formation, unilateral ureteral obstruction did not alter Pkd2-c-Halo expression or distribution. Finally, in a\n Pkd1<\/jats:italic>\n null background or in mice homozygous for\n Tulp3<\/jats:italic>\n R400W<\/jats:sup>\n , an allele identified in a patient with hepatorenal cystic disease, Pkd2-c-Halo failed to localize in cilia in kidney tubule epithelium.\n <\/jats:p>\n <\/jats:sec>\n \n Conclusions<\/jats:title>\n \n The Pkd2-c-Halo mouse and derived renal epithelial cell lines enable detection of endogenous polycystin-2\n in vivo<\/jats:italic>\n and\n in vitro<\/jats:italic>\n , allowing analysis of its localization, turnover under homeostatic conditions, after kidney injury, and in disease-relevant genetic backgrounds.\n <\/jats:p>\n <\/jats:sec>","DOI":"10.1681\/asn.0000001036","type":"journal-article","created":{"date-parts":[[2026,2,10]],"date-time":"2026-02-10T17:02:49Z","timestamp":1770742969000},"page":"1674-1688","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":0,"title":["A HaloTag Knock-In Resource for In Vivo and In Vitro Analysis of Endogenous Polycystin-2 Localization, Turnover, and Transport"],"prefix":"10.1681","volume":"37","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3796-0369","authenticated-orcid":false,"given":"Zhang","family":"Li","sequence":"first","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Courtney J.","family":"Haycraft","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5208-0530","authenticated-orcid":false,"given":"Mandy J.","family":"Croyle","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0008-6379-5863","authenticated-orcid":false,"given":"Daniel","family":"Hudson","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-3810-6487","authenticated-orcid":false,"given":"Yuan","family":"Yuan","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristin","family":"Simanyi","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6358-3579","authenticated-orcid":false,"given":"Hanan Chweih","family":"Vendrame","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3413-5208","authenticated-orcid":false,"given":"Jun","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alfiya Ibrahimbhai","family":"Kachwala","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yongjie","family":"Ma","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9084-5098","authenticated-orcid":false,"given":"John M.","family":"Parant","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Phillip","family":"Chumley","sequence":"additional","affiliation":[{"name":"Department of Medicine, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juling","family":"Zhou","sequence":"additional","affiliation":[{"name":"Department of Medicine, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michal","family":"Mrug","sequence":"additional","affiliation":[{"name":"Department of Medicine, University of Alabama at Birmingham, Birmingham, Alabama"},{"name":"Department of Veterans Affairs Medical Center, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8422-1147","authenticated-orcid":false,"given":"Stephen C.","family":"Parnell","sequence":"additional","affiliation":[{"name":"Department of Biochemistry and Molecular Biology, University of Kansas Medical Center, Kansas City, Kansas"},{"name":"Department of Internal Medicine, The Jared Grantham Kidney Institute, University of Kansas Medical Center, Kansas City, Kansas"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4619-4376","authenticated-orcid":false,"given":"Pamela V.","family":"Tran","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, The Jared Grantham Kidney Institute, University of Kansas Medical Center, Kansas City, Kansas"},{"name":"Department of Cell Biology and Physiology, University of Kansas Medical Center, Kansas City, Kansas"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hongjuan","family":"Gao","sequence":"additional","affiliation":[{"name":"Division of Nephrology, Department of Medicine, University of Maryland School of Medicine, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Feng","family":"Qian","sequence":"additional","affiliation":[{"name":"Division of Nephrology, Department of Medicine, University of Maryland School of Medicine, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0015-9228","authenticated-orcid":false,"given":"Patricia","family":"Outeda","sequence":"additional","affiliation":[{"name":"Division of Nephrology, Department of Medicine, University of Maryland School of Medicine, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4616-7669","authenticated-orcid":false,"given":"Darren P.","family":"Wallace","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, The Jared Grantham Kidney Institute, University of Kansas Medical Center, Kansas City, Kansas"},{"name":"Department of Cell Biology and Physiology, University of Kansas Medical Center, Kansas City, Kansas"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0812-7279","authenticated-orcid":false,"given":"Terry J.","family":"Watnick","sequence":"additional","affiliation":[{"name":"Division of Nephrology, Department of Medicine, University of Maryland School of Medicine, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0247-1944","authenticated-orcid":false,"given":"Bradley K.","family":"Yoder","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental and Integrative Biology, University of Alabama at Birmingham, Birmingham, Alabama"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"on behalf of the PKD Research Resource Consortium","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2026,2,10]]},"reference":[{"key":"B1-20260826","doi-asserted-by":"crossref","first-page":"321","DOI":"10.1146\/annurev.med.60.101707.125712","article-title":"Polycystic kidney disease","volume":"60","author":"Harris","year":"2009","journal-title":"Annu Rev Med."},{"issue":"6406","key":"B2-20260826","doi-asserted-by":"crossref","first-page":"eaat9819","DOI":"10.1126\/science.aat9819","article-title":"Structure of the human PKD1-PKD2 complex","volume":"361","author":"Su","year":"2018","journal-title":"Science."},{"issue":"3","key":"B3-20260826","doi-asserted-by":"crossref","first-page":"191","DOI":"10.1038\/ncb754","article-title":"Polycystin-2 is an intracellular calcium release channel","volume":"4","author":"Koulen","year":"2002","journal-title":"Nat Cell Biol."},{"issue":"580","key":"B4-20260826","doi-asserted-by":"crossref","first-page":"eaat7397","DOI":"10.1126\/scisignal.aat7397","article-title":"Polycystin 2 regulates mitochondrial Ca(2+) signaling, bioenergetics, and dynamics through mitofusin 2","volume":"12","author":"Kuo","year":"2019","journal-title":"Sci Signal."},{"issue":"2","key":"B5-20260826","doi-asserted-by":"crossref","first-page":"129","DOI":"10.1038\/ng1076","article-title":"Polycystins 1 and 2 mediate mechanosensation in the primary cilium of kidney cells","volume":"33","author":"Nauli","year":"2003","journal-title":"Nat Genet."},{"issue":"24","key":"B6-20260826","doi-asserted-by":"crossref","first-page":"4338","DOI":"10.1091\/mbc.e10-05-0407","article-title":"Polycystin-1 surface localization is stimulated by polycystin-2 and cleavage at the G protein-coupled receptor proteolytic site","volume":"21","author":"Chapin","year":"2010","journal-title":"Mol Biol Cell."},{"issue":"2","key":"B7-20260826","doi-asserted-by":"crossref","first-page":"278","DOI":"10.1681\/ASN.2008060564","article-title":"Characterization of PKD protein-positive exosome-like vesicles","volume":"20","author":"Hogan","year":"2009","journal-title":"J Am Soc Nephrol."},{"issue":"10","key":"B8-20260826","doi-asserted-by":"crossref","first-page":"1433","DOI":"10.1172\/JCI21753","article-title":"Mechanical stimuli induce cleavage and nuclear translocation of the polycystin-1 C terminus","volume":"114","author":"Chauvet","year":"2004","journal-title":"J Clin Invest."},{"issue":"9","key":"B9-20260826","doi-asserted-by":"crossref","first-page":"105158","DOI":"10.1016\/j.jbc.2023.105158","article-title":"Cleavage fragments of the C-terminal tail of polycystin-1 are regulated by oxidative stress and induce mitochondrial dysfunction","volume":"299","author":"Pellegrini","year":"2023","journal-title":"J Biol Chem."},{"issue":"10","key":"B10-20260826","doi-asserted-by":"crossref","first-page":"1459","DOI":"10.1172\/JCI5111","article-title":"Polycystin-1, the PKD1 gene product, is in a complex containing E-cadherin and the catenins","volume":"104","author":"Huan","year":"1999","journal-title":"J Clin Invest."},{"issue":"10","key":"B11-20260826","doi-asserted-by":"crossref","first-page":"2508","DOI":"10.1097\/01.ASN.0000029587.47950.25","article-title":"The polycystic kidney disease proteins, polycystin-1, polycystin-2, polaris, and cystin, are co-localized in renal cilia","volume":"13","author":"Yoder","year":"2002","journal-title":"J Am Soc Nephrol."},{"issue":"23","key":"B12-20260826","doi-asserted-by":"crossref","first-page":"20763","DOI":"10.1074\/jbc.M107788200","article-title":"Identification, characterization, and localization of a novel kidney polycystin-1-polycystin-2 complex","volume":"277","author":"Newby","year":"2002","journal-title":"J Biol Chem."},{"issue":"40","key":"B13-20260826","doi-asserted-by":"crossref","first-page":"28557","DOI":"10.1074\/jbc.274.40.28557","article-title":"Identification and characterization of polycystin-2, the PKD2 gene product","volume":"274","author":"Cai","year":"1999","journal-title":"J Biol Chem."},{"issue":"8","key":"B14-20260826","doi-asserted-by":"crossref","first-page":"e0289778","DOI":"10.1371\/journal.pone.0289778","article-title":"In vivo Polycystin-1 interactome using a novel Pkd1 knock-in mouse model","volume":"18","author":"Lin","year":"2023","journal-title":"PLoS One."},{"key":"B15-20260826","doi-asserted-by":"crossref","first-page":"109490","DOI":"10.1016\/j.cellsig.2019.109490","article-title":"Polycystin 2: a calcium channel, channel partner, and regulator of calcium homeostasis in ADPKD","volume":"66","author":"Brill","year":"2020","journal-title":"Cell Signal."},{"issue":"2","key":"B17-20260826","doi-asserted-by":"crossref","first-page":"607","DOI":"10.1172\/JCI76972","article-title":"Polycystin-1 maturation requires polycystin-2 in a dose-dependent manner","volume":"125","author":"Gainullin","year":"2015","journal-title":"J Clin Invest."},{"issue":"6815","key":"B18-20260826","doi-asserted-by":"crossref","first-page":"990","DOI":"10.1038\/35050128","article-title":"Co-assembly of polycystin-1 and -2 produces unique cation-permeable currents","volume":"408","author":"Hanaoka","year":"2000","journal-title":"Nature."},{"issue":"1","key":"B19-20260826","doi-asserted-by":"crossref","first-page":"2899","DOI":"10.1038\/s41467-025-57512-3","article-title":"Polycystins recruit cargo to distinct ciliary extracellular vesicle subtypes in C. elegans","volume":"16","author":"Nikonorova","year":"2025","journal-title":"Nat Commun."},{"issue":"6","key":"B20-20260826","doi-asserted-by":"crossref","first-page":"373","DOI":"10.1021\/cb800025k","article-title":"HaloTag: a novel protein labeling technology for cell imaging and protein analysis","volume":"3","author":"Los","year":"2008","journal-title":"ACS Chem Biol."},{"issue":"6","key":"B21-20260826","doi-asserted-by":"crossref","first-page":"975","DOI":"10.1021\/acs.bioconjchem.5b00191","article-title":"HaloTag technology: a versatile platform for biomedical applications","volume":"26","author":"England","year":"2015","journal-title":"Bioconjug Chem."},{"issue":"2","key":"B22-20260826","doi-asserted-by":"crossref","first-page":"200","DOI":"10.1039\/D3CC05536A","article-title":"Engineered fluorogenic HaloTag ligands for turn-on labelling in live cells","volume":"60","author":"Lampkin","year":"2023","journal-title":"Chem Commun (Camb)."},{"issue":"10","key":"B23-20260826","doi-asserted-by":"crossref","first-page":"987","DOI":"10.1038\/nmeth.4403","article-title":"A general method to fine-tune fluorophores for live-cell and in vivo imaging","volume":"14","author":"Grimm","year":"2017","journal-title":"Nat Methods."},{"issue":"34","key":"B24-20260826","doi-asserted-by":"crossref","first-page":"23963","DOI":"10.1021\/jacs.4c07080","article-title":"Chemigenetic far-red labels and Ca(2+) indicators optimized for photoacoustic imaging","volume":"146","author":"Cook","year":"2024","journal-title":"J Am Chem Soc."},{"issue":"12","key":"B25-20260826","doi-asserted-by":"crossref","first-page":"e202300022","DOI":"10.1002\/cbic.202300022","article-title":"HaloTag-Based reporters for fluorescence imaging and biosensing","volume":"24","author":"Cook","year":"2023","journal-title":"Chembiochem."},{"issue":"1","key":"B26-20260826","doi-asserted-by":"crossref","first-page":"e81","DOI":"10.1002\/cpph.81","article-title":"Targeted protein degradation phenotypic studies using HaloTag CRISPR\/Cas9 endogenous tagging coupled with HaloPROTAC3","volume":"91","author":"Caine","year":"2020","journal-title":"Curr Protoc Pharmacol."},{"issue":"5","key":"B27-20260826","doi-asserted-by":"crossref","first-page":"882","DOI":"10.1021\/acschembio.8b01016","article-title":"Rapid and reversible knockdown of endogenously tagged endosomal proteins via an optimized HaloPROTAC degrader","volume":"14","author":"Tovell","year":"2019","journal-title":"ACS Chem Biol."},{"issue":"8","key":"B28-20260826","doi-asserted-by":"crossref","first-page":"1831","DOI":"10.1021\/acschembio.5b00442","article-title":"HaloPROTACS: use of small molecule PROTACs to induce degradation of HaloTag fusion proteins","volume":"10","author":"Buckley","year":"2015","journal-title":"ACS Chem Biol."},{"issue":"10","key":"B29-20260826","doi-asserted-by":"crossref","first-page":"1411","DOI":"10.1021\/acs.accounts.2c00061","article-title":"Molecular spatiomics by proximity labeling","volume":"55","author":"Kang","year":"2022","journal-title":"Acc Chem Res."},{"issue":"2","key":"B30-20260826","doi-asserted-by":"crossref","first-page":"305","DOI":"10.1006\/dbio.2002.0597","article-title":"Efficient recombination in diverse tissues by a tamoxifen-inducible form of Cre: a tool for temporally regulated gene activation\/inactivation in the mouse","volume":"244","author":"Hayashi","year":"2002","journal-title":"Dev Biol."},{"issue":"9","key":"B31-20260826","doi-asserted-by":"crossref","first-page":"e12821","DOI":"10.1371\/journal.pone.0012821","article-title":"Pkd1 and Pkd2 are required for normal placental development","volume":"5","author":"Garcia-Gonzalez","year":"2010","journal-title":"PLoS One."},{"issue":"1","key":"B32-20260826","doi-asserted-by":"crossref","first-page":"8","DOI":"10.1186\/2046-2530-2-8","article-title":"An inducible CiliaGFP mouse model for in vivo visualization and analysis of cilia in live tissue","volume":"2","author":"O'Connor","year":"2013","journal-title":"Cilia."},{"issue":"9","key":"B33-20260826","doi-asserted-by":"crossref","first-page":"e7137","DOI":"10.1371\/journal.pone.0007137","article-title":"A novel mouse model reveals that polycystin-1 deficiency in ependyma and choroid plexus results in dysfunctional cilia and hydrocephalus","volume":"4","author":"Wodarczyk","year":"2009","journal-title":"PLoS One."},{"issue":"14","key":"B34-20260826","doi-asserted-by":"crossref","first-page":"2523","DOI":"10.1093\/hmg\/ddp147","article-title":"Renal injury is a third hit promoting rapid development of adult polycystic kidney disease","volume":"18","author":"Takakura","year":"2009","journal-title":"Hum Mol Genet."},{"issue":"4","key":"B35-20260826","doi-asserted-by":"crossref","first-page":"1493","DOI":"10.2353\/ajpath.2009.090227","article-title":"Pkd2 dosage influences cellular repair responses following ischemia-reperfusion injury","volume":"175","author":"Prasad","year":"2009","journal-title":"Am J Pathol."},{"issue":"18","key":"B36-20260826","doi-asserted-by":"crossref","first-page":"1586","DOI":"10.1016\/j.cub.2007.08.034","article-title":"Disruption of intraflagellar transport in adult mice leads to obesity and slow-onset cystic kidney disease","volume":"17","author":"Davenport","year":"2007","journal-title":"Curr Biol."},{"issue":"3","key":"B37-20260826","doi-asserted-by":"crossref","first-page":"834","DOI":"10.1093\/ndt\/gfm743","article-title":"Renal cilia display length alterations following tubular injury and are present early in epithelial repair","volume":"23","author":"Verghese","year":"2008","journal-title":"Nephrol Dial Transplant."},{"key":"B38-20260826","doi-asserted-by":"crossref","first-page":"1021037","DOI":"10.3389\/fgene.2022.1021037","article-title":"A pathogenic variant of TULP3 causes renal and hepatic fibrocystic disease","volume":"13","author":"Jafari Khamirani","year":"2022","journal-title":"Front Genet."},{"issue":"19","key":"B39-20260826","doi-asserted-by":"crossref","first-page":"2180","DOI":"10.1101\/gad.1966210","article-title":"TULP3 bridges the IFT-A complex and membrane phosphoinositides to promote trafficking of G protein-coupled receptors into primary cilia","volume":"24","author":"Mukhopadhyay","year":"2010","journal-title":"Genes Dev."},{"issue":"5","key":"B40-20260826","doi-asserted-by":"crossref","first-page":"790","DOI":"10.1016\/j.cub.2019.01.047","article-title":"Tulp3 regulates renal cystogenesis by trafficking of cystoproteins to cilia","volume":"29","author":"Hwang","year":"2019","journal-title":"Curr Biol."},{"key":"B41-20260826","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1186\/s13069-015-0024-y","article-title":"Primary cilia modulate balance of canonical and non-canonical Wnt signaling responses in the injured kidney","volume":"8","author":"Saito","year":"2015","journal-title":"Fibrogenesis Tissue Repair."},{"issue":"1","key":"B42-20260826","doi-asserted-by":"crossref","first-page":"ar1","DOI":"10.1091\/mbc.E24-09-0426","article-title":"A defined tubby domain beta-barrel surface region of TULP3 mediates ciliary trafficking of diverse cargoes","volume":"36","author":"Palicharla","year":"2025","journal-title":"Mol Biol Cell."},{"issue":"3","key":"B43-20260826","doi-asserted-by":"crossref","first-page":"743","DOI":"10.1083\/jcb.201607095","article-title":"Tubby family proteins are adapters for ciliary trafficking of integral membrane proteins","volume":"216","author":"Badgandi","year":"2017","journal-title":"J Cell Biol."},{"issue":"3","key":"B44-20260826","doi-asserted-by":"crossref","first-page":"ar18","DOI":"10.1091\/mbc.E22-10-0473","article-title":"Interactions between TULP3 tubby domain and ARL13B amphipathic helix promote lipidated protein transport to cilia","volume":"34","author":"Palicharla","year":"2023","journal-title":"Mol Biol Cell."},{"issue":"5","key":"B45-20260826","doi-asserted-by":"crossref","first-page":"803","DOI":"10.1016\/j.cub.2019.01.054","article-title":"Tulp3 is a ciliary trafficking gene that regulates polycystic kidney disease","volume":"29","author":"Legue","year":"2019","journal-title":"Curr Biol."},{"issue":"12","key":"B46-20260826","doi-asserted-by":"crossref","first-page":"5129","DOI":"10.1172\/JCI67273","article-title":"Altered trafficking and stability of polycystins underlie polycystic kidney disease","volume":"124","author":"Cai","year":"2014","journal-title":"J Clin Invest."}],"container-title":["Journal of the American Society of Nephrology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.1681\/ASN.0000001036","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T12:05:06Z","timestamp":1787745906000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.1681\/ASN.0000001036"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,2,10]]},"references-count":45,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2026]]}},"URL":"https:\/\/doi.org\/10.1681\/asn.0000001036","relation":{},"ISSN":["1046-6673","1533-3450"],"issn-type":[{"value":"1046-6673","type":"print"},{"value":"1533-3450","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,2,10]]},"assertion":[{"value":"2025-08-11","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-01-29","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}