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This process is disrupted in polycystic kidney disease (PKD), resulting in dilations of the nephron and renal cyst formation. In the course of exploring G-protein-coupled signaling in the Xenopus pronephric kidney, we discovered that loss of the G-protein \u03b1 subunit, Gnas, results in a PKD phenotype. Polycystin 1, one of the genes mutated in human PKD, encodes a protein resembling a G-protein-coupled receptor. Furthermore, deletion of the G-protein-binding domain present in the intracellular C terminus of polycystin 1 impacts functionality. A comprehensive analysis of all the G-protein \u03b1 subunits expressed in the Xenopus pronephric kidney demonstrates that polycystin 1 recruits a select subset of G-protein \u03b1 subunits and that their knockdown \u2013 as in the case of Gnas \u2013 results in a PKD phenotype. Mechanistically, the phenotype is caused by increased endogenous G-protein \u03b2\/\u03b3 signaling and can be reversed by pharmacological inhibitors as well as knocking down Gnb1. Together, our data support the hypothesis that G proteins are recruited to the intracellular domain of PKD1 and that this interaction is crucial for its function in the kidney.<\/jats:p>","DOI":"10.1242\/dev.158931","type":"journal-article","created":{"date-parts":[[2018,3,12]],"date-time":"2018-03-12T20:20:13Z","timestamp":1520886013000},"source":"Crossref","is-referenced-by-count":29,"title":["Polycystin 1 loss of function is directly linked to an imbalance in G-protein signaling in the kidney"],"prefix":"10.1242","volume":"145","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1359-9820","authenticated-orcid":false,"given":"Bo","family":"Zhang","sequence":"first","affiliation":[{"name":"Cleveland Clinic Foundation, Lerner Research Institute 1 , Department of Cellular and Molecular Medicine, 9500 Euclid Avenue\/NC10, Cleveland, OH 44195 , USA"},{"name":"LSU Health Sciences Center 2 , Department of Cell Biology and Anatomy, 1901 Perdido Street, New Orleans, LA 70112 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Uyen","family":"Tran","sequence":"additional","affiliation":[{"name":"Cleveland Clinic Foundation, Lerner Research Institute 1 , Department of Cellular and Molecular Medicine, 9500 Euclid Avenue\/NC10, Cleveland, OH 44195 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6440-7975","authenticated-orcid":false,"given":"Oliver","family":"Wessely","sequence":"additional","affiliation":[{"name":"Cleveland Clinic Foundation, Lerner Research Institute 1 , Department of Cellular and Molecular Medicine, 9500 Euclid Avenue\/NC10, Cleveland, OH 44195 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2018,3,22]]},"reference":[{"key":"2024061223515851100_DEV158931C1","doi-asserted-by":"publisher","first-page":"281","DOI":"10.1007\/978-3-319-51436-9_11","article-title":"Primary cilia in cystic kidney disease","volume":"60","author":"Avasthi","year":"2017","journal-title":"Results Probl. 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