{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T18:08:20Z","timestamp":1777486100830,"version":"3.51.4"},"reference-count":43,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2021,11,23]],"date-time":"2021-11-23T00:00:00Z","timestamp":1637625600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["18K08398"],"award-info":[{"award-number":["18K08398"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["20K08814"],"award-info":[{"award-number":["20K08814"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["20K17442"],"award-info":[{"award-number":["20K17442"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["21K08484"],"award-info":[{"award-number":["21K08484"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Oral Diseases"],"published-print":{"date-parts":[[2023,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>Cherubism is a genetic disorder characterised by bilateral jawbone deformation. The associated jawbone lesions regress after puberty, whereas severe cases require surgical treatment. Although several drugs have been tested, fundamental treatment strategies for cherubism have not been established. The effectiveness of imatinib has recently been reported; however, its pharmaceutical mechanism remains unclear. In this study, we tested the effects of imatinib using a cherubism mouse model.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      We used\n                      <jats:italic>Sh3bp2<\/jats:italic>\n                      P416R cherubism mutant mice, which exhibit systemic organ inflammation and osteopenia. The effects of imatinib were determined using primary bone marrow\u2010derived macrophages. Imatinib was administered intraperitoneally to the mice, and serum tumour necrosis factor\u2010\u03b1 (TNF\u03b1), organ inflammation and bone properties were examined.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      The cherubism mutant macrophages produced higher levels of TNF\u03b1 in response to lipopolysaccharide compared to wild\u2010type macrophages, and imatinib did not significantly suppress TNF\u03b1 production. Although imatinib suppressed osteoclast formation\n                      <jats:italic>in vitro<\/jats:italic>\n                      , administering it\n                      <jats:italic>in vivo<\/jats:italic>\n                      did not suppress organ inflammation and osteopenia.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      The\n                      <jats:italic>in vivo<\/jats:italic>\n                      administration of imatinib had a minimal therapeutic impact in cherubism mutant mice. To establish better pharmaceutical interventions, it is necessary to integrate new findings from murine models with clinical data from patients with a definitive diagnosis of cherubism.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/odi.14073","type":"journal-article","created":{"date-parts":[[2021,11,7]],"date-time":"2021-11-07T08:15:03Z","timestamp":1636272903000},"page":"1089-1101","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Imatinib has minimal effects on inflammatory and osteopenic phenotypes in a murine cherubism model"],"prefix":"10.1111","volume":"29","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0103-1621","authenticated-orcid":false,"given":"Tomoyuki","family":"Mukai","sequence":"first","affiliation":[{"name":"Department of Immunology and Molecular Genetics Kawasaki Medical School Kurashiki Okayama Japan"},{"name":"Department of Rheumatology Kawasaki Medical School Kurashiki Okayama Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takahiko","family":"Akagi","sequence":"additional","affiliation":[{"name":"Department of Rheumatology Kawasaki Medical School Kurashiki Okayama Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sumie","family":"Hiramatsu Asano","sequence":"additional","affiliation":[{"name":"Department of Rheumatology Kawasaki Medical School Kurashiki Okayama Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ikue","family":"Tosa","sequence":"additional","affiliation":[{"name":"Department of Oral Rehabilitation and Regenerative Medicine Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences Okayama Okayama Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mitsuaki","family":"Ono","sequence":"additional","affiliation":[{"name":"Department of Molecular Biology and Biochemistry Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences Okayama Okayama Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mizuho","family":"Kittaka","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences and Comprehensive Care Indiana University School of Dentistry Indianapolis Indiana USA"},{"name":"Indiana Center for Musculoskeletal Health Indiana University School of Medicine Indianapolis Indiana USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yasuyoshi","family":"Ueki","sequence":"additional","affiliation":[{"name":"Department of Biomedical Sciences and Comprehensive Care Indiana University School of Dentistry Indianapolis Indiana USA"},{"name":"Indiana Center for Musculoskeletal Health Indiana University School of Medicine Indianapolis Indiana USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ayano","family":"Yahagi","sequence":"additional","affiliation":[{"name":"Department of Immunology and Molecular 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