{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,12]],"date-time":"2026-09-12T11:46:13Z","timestamp":1789213573355,"version":"build-2803163510"},"reference-count":54,"publisher":"American Association for the Advancement of Science (AAAS)","issue":"19","content-domain":{"domain":["www.science.org"],"crossmark-restriction":true},"short-container-title":["Sci. Adv."],"published-print":{"date-parts":[[2025,5,9]]},"abstract":"<jats:p>\n            The successful secretion and assembly of subcommissural organ (SCO)\u2013spondin are crucial for Reissner Fiber (RF) formation and body axis straightening in zebrafish. However, the mechanisms underlying RF formation remain largely unknown. Here, we report that the C-mannosyltransferase\n            <jats:italic toggle=\"yes\">dpy19l1l<\/jats:italic>\n            (dumpy-19 like 1 like) is expressed in the spinal cord during zebrafish embryonic development. Mutation in\n            <jats:italic toggle=\"yes\">dpy19l1l<\/jats:italic>\n            resulted in idiopathic scoliosis (IS)\u2013like body axis curvature in the absence of muscle or cilia defects. URP2 expression was down-regulated in\n            <jats:italic toggle=\"yes\">\n              dpy19l1l\n              <jats:sup>\u2212\/\u2212<\/jats:sup>\n            <\/jats:italic>\n            mutants. Notably, RF formation was impaired in\n            <jats:italic toggle=\"yes\">\n              dpy19l1l\n              <jats:sup>\u2212\/\u2212<\/jats:sup>\n            <\/jats:italic>\n            mutants, and a similar phenotype was induced in wild-type embryos by injecting messenger RNA encoding a C-mannosylation catalytic site-mutated\n            <jats:italic toggle=\"yes\">dpy19l1l<\/jats:italic>\n            variant (E106A\n            <jats:italic toggle=\"yes\">mdpy19l1l<\/jats:italic>\n            ). Furthermore, E106A mDPY19L1L failed to glycosylate Flag-tagged SCO-spondin TSRs (thrombospondin type 1 repeats). Our findings suggest that DPY19L1L-mediated C-mannosylation of SCO-spondin TSRs promotes RF formation and URP2 induction, representing a critical supplementary mechanism for body axis straightening in zebrafish.\n          <\/jats:p>","DOI":"10.1126\/sciadv.adv2032","type":"journal-article","created":{"date-parts":[[2025,5,9]],"date-time":"2025-05-09T17:58:21Z","timestamp":1746813501000},"update-policy":"https:\/\/doi.org\/10.34133\/aaas_crossmark","source":"Crossref","is-referenced-by-count":9,"title":["C-mannosyltransferase DPY19L1L-mediated Reissner Fiber formation is critical for zebrafish (\n            <i>Danio rerio<\/i>\n            ) body axis straightening"],"prefix":"10.1126","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4478-4399","authenticated-orcid":true,"given":"Guiyou","family":"Tian","sequence":"first","affiliation":[{"name":"Jiangxi Provincial Key Laboratory of Synthetic Pharmaceutical Chemistry, School of Geography and Environmental Engineering, Gannan Normal University, Ganzhou 341000, China."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-9764-3793","authenticated-orcid":true,"given":"Lirong","family":"Huang","sequence":"additional","affiliation":[{"name":"Jiangxi Provincial Key Laboratory of Synthetic Pharmaceutical Chemistry, School of Geography and Environmental Engineering, Gannan Normal University, Ganzhou 341000, China."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7279-2391","authenticated-orcid":true,"given":"Zhaopeng","family":"Xu","sequence":"additional","affiliation":[{"name":"Jiangxi Provincial Key Laboratory of Synthetic Pharmaceutical Chemistry, School of Geography and Environmental Engineering, Gannan Normal University, Ganzhou 341000, China."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-7861-5287","authenticated-orcid":true,"given":"Chen","family":"Lu","sequence":"additional","affiliation":[{"name":"Jiangxi Provincial Key Laboratory of Synthetic Pharmaceutical Chemistry, School of Geography and Environmental Engineering, Gannan Normal University, Ganzhou 341000, China."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wei","family":"Yuan","sequence":"additional","affiliation":[{"name":"Jiangxi Provincial Key Laboratory of Synthetic Pharmaceutical Chemistry, School of Geography and Environmental Engineering, Gannan Normal University, Ganzhou 341000, China."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-6997-9360","authenticated-orcid":true,"given":"Yulin","family":"Wu","sequence":"additional","affiliation":[{"name":"Jiangxi Provincial Key Laboratory of Synthetic Pharmaceutical Chemistry, School of 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