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In the kidney, the pathogenic mechanisms underlying the program of altered cell and tubule morphology are obscured by secondary effects of cyst expansion. Here, we developed a new 3D tubuloid system to isolate the rapid changes in protein localization and gene expression that correlate with altered cell and tubule morphology during cyst initiation. Mouse renal tubule fragments were pulsed with a cell differentiation cocktail including glial-derived neurotrophic factor (GDNF) to yield collecting duct-like tubuloid structures with appropriate polarity, primary cilia, and gene expression. Using the 3D tubuloid model with an inducible Pkd2 knockout system allowed the tracking of morphological, protein, and genetic changes during cyst formation. Within hours of inactivation of Pkd2 and loss of polycystin-2, we observed significant progression in tubuloid to cyst morphology that correlated with 35 differentially expressed genes, many related to cell junctions, matrix interactions, and cell morphology previously implicated in cystogenesis.<\/jats:p>\n               <jats:p>This article has an associated First Person interview with the first author of the paper.<\/jats:p>","DOI":"10.1242\/jcs.249557","type":"journal-article","created":{"date-parts":[[2020,6,8]],"date-time":"2020-06-08T13:54:37Z","timestamp":1591624477000},"source":"Crossref","is-referenced-by-count":14,"title":["GDNF drives rapid tubule morphogenesis in a novel 3D <i>in vitro<\/i> model for ADPKD"],"prefix":"10.1242","volume":"133","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4604-7907","authenticated-orcid":false,"given":"Eryn E.","family":"Dixon","sequence":"first","affiliation":[{"name":"University of Maryland School of Medicine 1 , Department of Physiology, Baltimore, MD 21201 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Demetrios S.","family":"Maxim","sequence":"additional","affiliation":[{"name":"University of Maryland School of Medicine 1 , Department of Physiology, Baltimore, MD 21201 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6348-7931","authenticated-orcid":false,"given":"Victoria L.","family":"Halperin Kuhns","sequence":"additional","affiliation":[{"name":"University of Maryland School of Medicine 1 , Department of Physiology, Baltimore, MD 21201 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Allison C.","family":"Lane-Harris","sequence":"additional","affiliation":[{"name":"University of Maryland School of Medicine 1 , Department of Physiology, Baltimore, MD 21201 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0015-9228","authenticated-orcid":false,"given":"Patricia","family":"Outeda","sequence":"additional","affiliation":[{"name":"University of Maryland School of Medicine 2 , Department of Medicine, Baltimore, MD 21201 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1964-0740","authenticated-orcid":false,"given":"Andrew J.","family":"Ewald","sequence":"additional","affiliation":[{"name":"Johns Hopkins University School of Medicine 3 , Department of Cell Biology, Baltimore, MD 21205 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0812-7279","authenticated-orcid":false,"given":"Terry J.","family":"Watnick","sequence":"additional","affiliation":[{"name":"University of Maryland School of Medicine 2 , Department of Medicine, Baltimore, MD 21201 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0719-8705","authenticated-orcid":false,"given":"Paul A.","family":"Welling","sequence":"additional","affiliation":[{"name":"Johns Hopkins University School of Medicine 4 , Departments of Medicine and Physiology, Baltimore , MD 21205 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9514-2180","authenticated-orcid":false,"given":"Owen M.","family":"Woodward","sequence":"additional","affiliation":[{"name":"University of Maryland School of Medicine 1 , Department of Physiology, Baltimore, MD 21201 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2020,7,16]]},"reference":[{"key":"2024080908361348800_JCS249557C1","doi-asserted-by":"publisher","first-page":"2081","DOI":"10.1681\/ASN.2014121192","article-title":"The future of polycystic kidney disease research\u2014as seen by the 12 Kaplan Awardees","volume":"26","author":"Antignac","year":"2015","journal-title":"J. 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