{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,24]],"date-time":"2026-09-24T16:21:33Z","timestamp":1790266893275,"version":"4.1.0"},"reference-count":30,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2023,12,25]],"date-time":"2023-12-25T00:00:00Z","timestamp":1703462400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100004948","name":"Astellas Pharma","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004948","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002973","name":"Daiichi-Sankyo","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100002973","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100015993","name":"Kowa Company","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100015993","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["febs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["FEBS Open Bio"],"published-print":{"date-parts":[[2024,2]]},"abstract":"<jats:p>\n                    Congenital fibroblast growth factor 23 (FGF23)\u2010related hypophosphatemic rickets\/osteomalacia is a rare bone metabolism disorder characterized by hypophosphatemia and caused by genetic abnormalities that result in excessive secretion of FGF23.\n                    <jats:italic>Hyp<\/jats:italic>\n                    mice are a model of X\u2010linked hypophosphatemia (XLH) caused by deletion of the\n                    <jats:italic>PHEX<\/jats:italic>\n                    gene and excessive production of FGF23. The purpose of this study was to investigate the potential of TM5614 as a therapeutic agent for the treatment of congenital FGF23\u2010related hypophosphatemic rickets and osteomalacia in humans by administering TM5614 to\n                    <jats:italic>Hyp<\/jats:italic>\n                    mice and examining its curative effect on hypophosphatemia. After a single oral administration of TM5614 10\u2009mg\u00b7kg\n                    <jats:sup>\u22121<\/jats:sup>\n                    to female\n                    <jats:italic>Hyp<\/jats:italic>\n                    mice starting at 17\u2009weeks of age, the serum phosphate concentration increased with a peak at 6\u2009h after administration. ELISA confirmed that TM5614 administration decreased the intact FGF23 concentration in the blood. Expression of 25\u2010hydroxyvitamin D\u20101\u03b1\u2010hydroxylase protein encoded by\n                    <jats:italic>Cyp27b1<\/jats:italic>\n                    mRNA in the kidney was suppressed in\n                    <jats:italic>Hyp<\/jats:italic>\n                    mice, and treatment with 10\u2009mg\u00b7kg\n                    <jats:sup>\u22121<\/jats:sup>\n                    of TM5614 normalized the expression of 25\u2010hydroxyvitamin D\u20101\u03b1\u2010hydroxylase protein and\n                    <jats:italic>Cyp27b1<\/jats:italic>\n                    mRNA in the kidneys of these mice. Our data indicate that oral administration of TM5614 ameliorates hypophosphatemia in\n                    <jats:italic>Hyp<\/jats:italic>\n                    mice, suggesting that TM5614 may be an effective treatment for congenital FGF23\u2010related hypophosphatemic rickets and osteomalacia.\n                  <\/jats:p>","DOI":"10.1002\/2211-5463.13745","type":"journal-article","created":{"date-parts":[[2023,12,5]],"date-time":"2023-12-05T03:34:42Z","timestamp":1701747282000},"page":"290-299","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["A\n                    <scp>PAI<\/scp>\n                    \u20101 antagonist ameliorates hypophosphatemia in the\n                    <i>Hyp<\/i>\n                    vitamin D\u2010resistant rickets model mouse"],"prefix":"10.1002","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0009-0008-6078-064X","authenticated-orcid":false,"given":"Cheng","family":"Qian","sequence":"first","affiliation":[{"name":"Department of Orthopedics Surgery Tokyo Medical and Dental University  Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nobuaki","family":"Ito","sequence":"additional","affiliation":[{"name":"Division of Nephrology and Endocrinology The University of Tokyo Hospital  Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kunikazu","family":"Tsuji","sequence":"additional","affiliation":[{"name":"Department of Orthopedics Surgery Tokyo Medical and Dental University  Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6227-5910","authenticated-orcid":false,"given":"Shingo","family":"Sato","sequence":"additional","affiliation":[{"name":"Department of Orthopedics Surgery Tokyo Medical and Dental 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