{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T01:38:15Z","timestamp":1791509895402,"version":"4.3.4"},"reference-count":91,"publisher":"The Company of Biologists","issue":"5","license":[{"start":{"date-parts":[[2020,9,3]],"date-time":"2020-09-03T00:00:00Z","timestamp":1599091200000},"content-version":"vor","delay-in-days":491,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100003725","name":"National Research Foundation of Korea","doi-asserted-by":"publisher","award":["NRF-2011-0023507"],"award-info":[{"award-number":["NRF-2011-0023507"]}],"id":[{"id":"10.13039\/501100003725","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100008783","name":"National Research Council of Science & Technology","doi-asserted-by":"publisher","award":["CRC-15-04-KIST"],"award-info":[{"award-number":["CRC-15-04-KIST"]}],"id":[{"id":"10.13039\/501100008783","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003621","name":"Ministry of Science, ICT and Future Planning","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003621","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003715","name":"Korea Research Institute of Bioscience and Biotechnology","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003715","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2019,5,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>DYRK1A is a major causative gene in Down syndrome (DS). Reduced incidence of solid tumors such as neuroblastoma in DS patients and increased vascular anomalies in DS fetuses suggest a potential role of DYRK1A in angiogenic processes, but in vivo evidence is still scarce. Here, we used zebrafish dyrk1aa mutant embryos to understand DYRK1A function in cerebral vasculature formation. Zebrafish dyrk1aa mutants exhibited cerebral hemorrhage and defects in angiogenesis of central arteries in the developing hindbrain. Such phenotypes were rescued by wild-type dyrk1aa mRNA, but not by a kinase-dead form, indicating the importance of DYRK1A kinase activity. Chemical screening using a bioactive small molecule library identified a calcium chelator, EGTA, as one of the hits that most robustly rescued the hemorrhage. Vascular defects of mutants were also rescued by independent modulation of calcium signaling by FK506. Furthermore, the transcriptomic analyses supported the alterations of calcium signaling networks in dyrk1aa mutants. Together, our results suggest that DYRK1A plays an essential role in angiogenesis and in maintenance of the developing cerebral vasculature via regulation of calcium signaling, which may have therapeutic potential for DYRK1A-related vascular diseases.<\/jats:p>","DOI":"10.1242\/dmm.037044","type":"journal-article","created":{"date-parts":[[2019,5,1]],"date-time":"2019-05-01T08:15:13Z","timestamp":1556698513000},"source":"Crossref","is-referenced-by-count":26,"title":["Vascular defects of\n                    <i>DYRK1A<\/i>\n                    knockouts are ameliorated by modulating calcium signaling in zebrafish"],"prefix":"10.1242","volume":"12","author":[{"given":"Hyun-Ju","family":"Cho","sequence":"first","affiliation":[{"name":"Disease Target Structure Research Center 1 , Korea Research Institute of Bioscience and Biotechnology , 125 Gwahak-ro, Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"KRIBB School, University of Science and Technology 2 , 125 Gwahak-ro , Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"Korea Institute of Science and Technology 3 Dementia DTC R&D Convergence Program , , Hwarang-ro 14-gil 5, Seongbuk-gu, Seoul, 02792 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jae-Geun","family":"Lee","sequence":"additional","affiliation":[{"name":"Disease Target Structure Research Center 1 , Korea Research Institute of Bioscience and Biotechnology , 125 Gwahak-ro, Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"KRIBB School, University of Science and Technology 2 , 125 Gwahak-ro , Yuseong-gu, Daejeon, 34141 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jong-Hwan","family":"Kim","sequence":"additional","affiliation":[{"name":"KRIBB School, University of Science and Technology 2 , 125 Gwahak-ro , Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"Genome Editing Research Center, Korea Research Institute of Bioscience and Biotechnology 4 , 125 Gwahak-ro, Yuseong-gu, Daejeon, 34141 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seon-Young","family":"Kim","sequence":"additional","affiliation":[{"name":"KRIBB School, University of Science and Technology 2 , 125 Gwahak-ro , Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"Genome Editing Research Center, Korea Research Institute of Bioscience and Biotechnology 4 , 125 Gwahak-ro, Yuseong-gu, Daejeon, 34141 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yang Hoon","family":"Huh","sequence":"additional","affiliation":[{"name":"Electron Microscopy Research Center, Korea Basic Science Institute 5 , 162 Yeongudanji-ro, Ochang-eup, Cheongwon-gu, Cheongju-si, Chungcheongbuk-do, 28119 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hyo-Jeong","family":"Kim","sequence":"additional","affiliation":[{"name":"Electron Microscopy Research Center, Korea Basic Science Institute 5 , 162 Yeongudanji-ro, Ochang-eup, Cheongwon-gu, Cheongju-si, Chungcheongbuk-do, 28119 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kyu-Sun","family":"Lee","sequence":"additional","affiliation":[{"name":"KRIBB School, University of Science and Technology 2 , 125 Gwahak-ro , Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"Hazards Monitoring BNT Research Center 6 , Korea Research Institute of Bioscience and Biotechnology , 125 Gwahak-ro, Yuseong-gu, Daejeon, 34141 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kweon","family":"Yu","sequence":"additional","affiliation":[{"name":"Disease Target Structure Research Center 1 , Korea Research Institute of Bioscience and Biotechnology , 125 Gwahak-ro, Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"KRIBB School, University of Science and Technology 2 , 125 Gwahak-ro , Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"Korea Institute of Science and Technology 3 Dementia DTC R&D Convergence Program , , Hwarang-ro 14-gil 5, Seongbuk-gu, Seoul, 02792 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8491-4429","authenticated-orcid":false,"given":"Jeong-Soo","family":"Lee","sequence":"additional","affiliation":[{"name":"Disease Target Structure Research Center 1 , Korea Research Institute of Bioscience and Biotechnology , 125 Gwahak-ro, Yuseong-gu, Daejeon, 34141 , Republic of Korea"},{"name":"Korea Institute of Science and Technology 3 Dementia DTC R&D Convergence Program , , Hwarang-ro 14-gil 5, Seongbuk-gu, Seoul, 02792 , Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2019,5,23]]},"reference":[{"key":"2024061302185229700_DMM037044C1","doi-asserted-by":"publisher","first-page":"251","DOI":"10.4103\/0253-7613.132152","article-title":"Assessment methods for 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