{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,3]],"date-time":"2026-11-03T07:56:37Z","timestamp":1793692597478,"version":"4.5.1"},"reference-count":52,"publisher":"National Academy of Sciences","issue":"43","license":[{"start":{"date-parts":[[2024,10,15]],"date-time":"2024-10-15T00:00:00Z","timestamp":1728950400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000062","name":"HHS | NIH | National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"publisher","award":["UC2DK126024"],"award-info":[{"award-number":["UC2DK126024"]}],"id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000062","name":"HHS | NIH | National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"publisher","award":["2R01DK103740"],"award-info":[{"award-number":["2R01DK103740"]}],"id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000062","name":"HHS | NIH | National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"publisher","award":["1U54DK137332"],"award-info":[{"award-number":["1U54DK137332"]}],"id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000005","name":"U.S. Department of Defense","doi-asserted-by":"publisher","award":["W81XWH-20-1-0198"],"award-info":[{"award-number":["W81XWH-20-1-0198"]}],"id":[{"id":"10.13039\/100000005","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["www.pnas.org"],"crossmark-restriction":true},"short-container-title":["Proc. Natl. Acad. Sci. U.S.A."],"published-print":{"date-parts":[[2024,10,22]]},"abstract":"<jats:p>\n                    Autosomal dominant polycystic kidney disease (ADPKD) is the most common hereditary kidney disease and causes significant morbidity, ultimately leading to kidney failure. PKD pathogenesis is characterized by complex and dynamic alterations in multiple cell types during disease progression, hampering a deeper understanding of disease mechanism and the development of therapeutic approaches. Here, we generate a single-nucleus multimodal atlas of an orthologous mouse PKD model at early, mid, and late timepoints, consisting of 125,434 single-nucleus transcriptomic and epigenetic multiomes. We catalog differentially expressed genes and activated epigenetic regions in each cell type during PKD progression, characterizing cell-type-specific responses to\n                    <jats:italic>Pkd1<\/jats:italic>\n                    deletion. We describe heterogeneous, atypical collecting duct cells as well as proximal tubular cells that constitute cyst epithelia in PKD. The transcriptional regulation of the cyst lining cell marker GPRC5A is conserved between mouse and human PKD cystic epithelia, suggesting shared gene regulatory pathways. Our single-nucleus multiomic analysis of mouse PKD provides a foundation to understand the earliest changes molecular deregulation in a mouse model of PKD at a single-cell resolution.\n                  <\/jats:p>","DOI":"10.1073\/pnas.2410830121","type":"journal-article","created":{"date-parts":[[2024,10,15]],"date-time":"2024-10-15T13:32:41Z","timestamp":1728999161000},"update-policy":"https:\/\/doi.org\/10.1073\/pnas.cm10313","source":"Crossref","is-referenced-by-count":26,"title":["Multiomics profiling of mouse polycystic kidney disease progression at a single-cell resolution"],"prefix":"10.1073","volume":"121","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0358-9442","authenticated-orcid":false,"given":"Yoshiharu","family":"Muto","sequence":"first","affiliation":[{"name":"Division of Nephrology, Department of Internal Medicine, University of Texas Southwestern Medical Center, Dallas, TX 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63110"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Owen M.","family":"Woodward","sequence":"additional","affiliation":[{"name":"Department of Physiology, University of Maryland School of Medicine, Baltimore, MD 21201"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Patricia","family":"Outeda","sequence":"additional","affiliation":[{"name":"Department of Medicine, University of Maryland School of Medicine, Baltimore, MD 21201"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tao","family":"Cheng","sequence":"additional","affiliation":[{"name":"Division of Nephrology, Department of Medicine, Washington University in St. Louis, St. Louis, MO 63110"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8129-7464","authenticated-orcid":false,"given":"Moe R.","family":"Mahjoub","sequence":"additional","affiliation":[{"name":"Division of Nephrology, Department of Medicine, Washington University in St. Louis, St. Louis, MO 63110"},{"name":"Department of Cell Biology and Physiology, Washington University in St. Louis, St. Louis, MO 63110"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Terry J.","family":"Watnick","sequence":"additional","affiliation":[{"name":"Department of Medicine, University of Maryland School of Medicine, Baltimore, MD 21201"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6420-8703","authenticated-orcid":false,"given":"Benjamin D.","family":"Humphreys","sequence":"additional","affiliation":[{"name":"Division of Nephrology, Department of Medicine, Washington University in St. Louis, St. Louis, MO 63110"},{"name":"Department of Developmental Biology, Washington University in St. Louis, St. Louis, MO 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