{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,21]],"date-time":"2026-10-21T16:47:30Z","timestamp":1792601250179,"version":"4.5.0"},"reference-count":27,"publisher":"Oxford University Press (OUP)","issue":"9","license":[{"start":{"date-parts":[[2023,6,21]],"date-time":"2023-06-21T00:00:00Z","timestamp":1687305600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["asbmr.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["JBMR Plus"],"published-print":{"date-parts":[[2023,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    The fracture experience of children and adolescents with osteogenesis imperfecta (OI) type 1 is not well described in the literature. We present data on symptomatic long bones and axial skeleton fractures of all patients aged 0 to 18\u2009years with OI type 1 seen at a specialized bone clinic in Western Australia in the period 2008 to 2020 using a retrospective chart review method. The cohort consisted of 44 patients (21 males, 23 females). Median (interquartile range [IQR]) age was 11.3 (6.2 to 17) years, giving a total of 520 patient\u2010years in the study during which 197 fractures were experienced. The mean fracture rate was 379 fractures per 1000 patient\u2010years (95% confidence interval [CI]: 310 to 440); however, the experience for fractures varied from \u22641 fracture in 23% (\n                    <jats:italic>n<\/jats:italic>\n                    \u2009=\u200910) to two to 20 in 77% (\n                    <jats:italic>n<\/jats:italic>\n                    \u2009=\u200934) of the cohort. Twenty\u2010one patients (48.5%) received bisphosphonates during the study period. In logistic regression, age, but not sex or family history of OI, was a significant predictor of fracture risk. The highest total fracture rate was observed in the age group 0 to &lt;3\u2009years at 469 fractures\/1000 patient\u2010years, which declined to 140 fractures\/1000 patient\u2010years in the age group 15 to 18\u2009years. The lower limbs were the site of 49.7% of all fractures. The highest rate for lower limb fracture was in the age group 0 to &lt;3\u2009years at 331 fractures\/1000 patient\u2010years, decreasing to 0 fractures\/1000 patient\u2010years in the age group 15 to 18\u2009years. Upper limb fracture rates increased from 100 fractures\/1000 patient\u2010years in the 0 to &lt;3\u2009years age group to 307 fractures\/1000 patient\u2010years in the 9 to &lt;12\u2009years age group and then declining to 70 fractures\/1000\u2009years in the 15 to 18\u2009years age group. In pediatric patients with OI type 1, fracture risk is highest in early life, especially in the lower limbs. Multidisciplinary care of children with OI should have a particular focus on strategies to prevent these fractures. \u00a9 2023 The Authors.\n                    <jats:italic>JBMR Plus<\/jats:italic>\n                    published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research.\n                  <\/jats:p>","DOI":"10.1002\/jbm4.10782","type":"journal-article","created":{"date-parts":[[2023,6,23]],"date-time":"2023-06-23T12:28:51Z","timestamp":1687523331000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["The Natural History of Symptomatic Fractures in Children and Adolescents with Osteogenesis Imperfecta Type 1: A Cohort Study from Western Australia"],"prefix":"10.1093","volume":"7","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4323-7441","authenticated-orcid":false,"given":"Kiranjit K","family":"Joshi","sequence":"first","affiliation":[{"name":"University of Western Australia  Perth WA Australia"},{"name":"Perth Children's Hospital Department of Endocrinology and Diabetes  Perth WA Australia"},{"name":"Western Australian Bone Research Collaboration  Perth WA Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aris","family":"Siafarikas","sequence":"additional","affiliation":[{"name":"University of Western Australia  Perth WA Australia"},{"name":"Perth Children's Hospital Department of Endocrinology and Diabetes  Perth WA Australia"},{"name":"Western Australian Bone Research Collaboration  Perth WA Australia"},{"name":"Institute for Health Research University of Notre Dame Australia  Fremantle WA Australia"},{"name":"Exercise Medicine Research Institute Edith Cowan University  Perth WA Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard","family":"Prince","sequence":"additional","affiliation":[{"name":"University of Western Australia  Perth WA Australia"},{"name":"Western Australian Bone 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