{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,25]],"date-time":"2026-10-25T02:59:45Z","timestamp":1792897185790,"version":"4.5.0"},"reference-count":30,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2018,10,24]],"date-time":"2018-10-24T00:00:00Z","timestamp":1540339200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"},{"start":{"date-parts":[[2018,11,29]],"date-time":"2018-11-29T00:00:00Z","timestamp":1543449600000},"content-version":"vor","delay-in-days":36,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["asbmr.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2019,2,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:sec>\n                  <jats:title>\u2003<\/jats:title>\n                  <jats:p>Osteogenesis imperfecta (OI) is a heritable bone fragility disorder that is usually caused by mutations affecting collagen type I encoding genes. Recent studies in mouse models of recessive OI, Crtap-\/- mice, and dominant OI, +\/G610C mice, found that application of a transforming growth factor beta (TGF-\u03b2) neutralizing antibody 1D11 rescues the bone phenotype. In the present study, we investigated TGF-\u03b2 signaling in a mouse model of severe dominant OI with a high incidence of spontaneous fractures, Col1a1Jrt\/+ mice, and the effect of TGF-\u03b2 neutralizing antibody 1D11 on bone phenotype in 8-week-old mice. Col1a1Jrt\/+ mice had elevated TGF-\u03b2 signaling in bone tissue. Treatment of Col1a1Jrt\/+ mice with 1D11 was associated with increased bone length but had no significant effect on bone mass or bone mechanical properties, and no significant treatment-associated differences in serum markers of bone formation (alkaline phosphatase activity) or resorption (tartrate-resistant acid phosphatase) were found. Our data thus indicate that the TGF-\u03b2 neutralizing antibody 1D11 is not effective in a mouse model of dominant OI with a high incidence of spontaneous fractures. \u00a9 2018 American Society for Bone and Mineral Research.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1002\/jbmr.3617","type":"journal-article","created":{"date-parts":[[2018,10,24]],"date-time":"2018-10-24T21:13:02Z","timestamp":1540415582000},"page":"207-214","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":54,"title":["Effect of Anti-TGF-\u03b2 Treatment in a Mouse Model of Severe Osteogenesis Imperfecta"],"prefix":"10.1093","volume":"34","author":[{"given":"Josephine T","family":"Tauer","sequence":"first","affiliation":[{"name":"Shriners Hospital for Children-CanadaMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sami","family":"Abdullah","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children-CanadaMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank","family":"Rauch","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children-CanadaMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2018,10,24]]},"reference":[{"issue":"12","key":"2024021014514588600_jbmr3617-bib-0001","doi-asserted-by":"crossref","first-page":"3607","DOI":"10.1007\/s00198-016-3709-1","article-title":"DNA sequence analysis in 598 individuals with a clinical diagnosis of osteogenesis imperfecta: diagnostic yield and mutation spectrum","volume":"27","author":"Bardai","year":"2016","journal-title":"Osteoporos Int"},{"issue":"11","key":"2024021014514588600_jbmr3617-bib-0002","doi-asserted-by":"crossref","first-page":"2788","DOI":"10.1021\/acsbiomaterials.6b00300","article-title":"Electron microscopy reveals structural and chemical changes at the nanometer scale in the 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