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While PKD1 mutant mouse models have provided mechanistic insights, PKD1 heterozygous mice fail to replicate the early cystogenesis observed in human patients. To address this gap, we conducted a longitudinal study using PKD1 heterozygous cynomolgus monkeys. Serial renal ultrasonography from birth to five years of age\u2014corresponding to human childhood\u2014revealed progressive cyst development. Remarkably, a subset of cysts, particularly smaller ones, exhibited spontaneous regression over time. This phenomenon was also observed in PKD1 mosaic monkeys harboring mixed variant patterns. In contrast, monkeys with biallelic PKD1 loss-of-function variants developed severe cystic disease, kidney enlargement, hepatic cysts, and elevated serum creatinine, resembling the clinical features of advanced ADPKD. These findings demonstrate that early renal cysts may possess intrinsic plasticity, challenging the conventional view of ADPKD as a relentlessly progressive disorder. Our results suggest that the early stages of cystogenesis represent a potential therapeutic window for intervention, in which cyst regression may be promoted. The PKD1 heterozygous monkey model thus provides a valuable platform for studying the developmental dynamics of ADPKD and for evaluating novel therapeutic approaches targeting early cystic changes.<\/jats:p>","DOI":"10.1093\/hmg\/ddag011","type":"journal-article","created":{"date-parts":[[2026,3,17]],"date-time":"2026-03-17T18:09:01Z","timestamp":1773770941000},"source":"Crossref","is-referenced-by-count":0,"title":["Reversible cystogenesis in juvenile primate ADPKD models: evidence from\n                    <i>PKD1<\/i>\n                    heterozygous monkeys"],"prefix":"10.1093","volume":"35","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8662-4636","authenticated-orcid":false,"given":"Shoma","family":"Matsumoto","sequence":"first","affiliation":[{"name":"Department of Stem Cells and Human Disease Models, Research Center for Animal Life Science, Shiga University of Medical Science , Seta Tsukinowa-cho, Otsu, Shiga 520-2192 ,","place":["Japan"]},{"name":"Shoma Matsumoto, Department of Creative Destruction Biology, Premium Research Institute for Human Metaverse Medicine (WPI-PRIMe), The University of Osaka, The Center of Medical Innovation and Translational Research #305 , 2-2, Yamadaoka, Suita, Osaka 565-0871, Japan, Tel.: +81-6-6879-5111, Email: matsumoto.shoma.prime@osaka-u.ac.jp"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Toshifumi","family":"Morimura","sequence":"additional","affiliation":[{"name":"Department of Stem Cells and Human Disease Models, Research Center for Animal Life Science, Shiga University of Medical Science , Seta Tsukinowa-cho, Otsu, Shiga 520-2192 ,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kenichi","family":"Kobayashi","sequence":"additional","affiliation":[{"name":"Department of Stem Cells and Human Disease Models, Research Center for Animal Life Science, Shiga University of Medical Science , Seta Tsukinowa-cho, Otsu, Shiga 520-2192 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891-1394"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Koji","family":"Fukuda","sequence":"additional","affiliation":[{"name":"Shin Nippon Biomedical Laboratories, Ltd. , 2438, Miyanoura-cho, Kagoshima, Kagoshima ,","place":["Japan, 891-1394"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Teppei","family":"Iwakiri","sequence":"additional","affiliation":[{"name":"Shin Nippon Biomedical Laboratories, Ltd. , 2438, Miyanoura-cho, Kagoshima, Kagoshima ,","place":["Japan, 891-1394"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kazuhiko","family":"Nozaki","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Shiga University of Medical Science , Seta Tsukinowa-cho, Otsu, Shiga 520-2192,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Akihiro","family":"Kawauchi","sequence":"additional","affiliation":[{"name":"Department of Urology, Shiga University of Medical Science , Seta Tsukinowa-cho, Otsu, Shiga 520-2192 ,","place":["Japan"]},{"name":"Otsu City Hospital , 2-9-9, Motomiya, Otsu, Shiga 520-0804,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Susumu","family":"Kageyama","sequence":"additional","affiliation":[{"name":"Department of Urology, Shiga University of Medical Science , Seta Tsukinowa-cho, Otsu, Shiga 520-2192 ,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shinji","family":"Kume","sequence":"additional","affiliation":[{"name":"Department of Medicine, Shiga University of Medical Science , Seta Tsukinowa-cho, Otsu, Shiga 520-2192,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Saori","family":"Nishio","sequence":"additional","affiliation":[{"name":"Department of Hemodialysis and Apheresis, Hokkaido University Hospital , Kita14, Nishi5, Kita-ku, Sapporo , Hokkaido 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