{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,2]],"date-time":"2026-07-02T03:52:02Z","timestamp":1782964322880,"version":"3.54.5"},"reference-count":26,"publisher":"Oxford University Press (OUP)","issue":"12","license":[{"start":{"date-parts":[[2019,7,29]],"date-time":"2019-07-29T00:00:00Z","timestamp":1564358400000},"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":[[2019,8,26]],"date-time":"2019-08-26T00:00:00Z","timestamp":1566777600000},"content-version":"vor","delay-in-days":28,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["asbmr.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2019,12,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Intravenous cyclical bisphosphonates are widely used to treat children with moderate to severe osteogenesis imperfecta (OI). Bisphosphonates are often discontinued when growth is completed, but subsequent skeletal changes have not been studied in detail. We assessed 31 patients (22 females) with OI who had started intravenous bisphosphonates (either pamidronate or zoledronic acid) before 13\u2009years of age, were treated for at least 2\u2009years (range 4.7\u201315.7\u2009years), and discontinued treatment after completion of growth, when their age ranged from 13.4 to 20.0\u2009years (mean 16.4\u2009years). At 4\u2009years after treatment discontinuation, lumbar spine areal bone mineral density (BMD) had increased by 4% (p\u2009&amp;lt;\u20090.05). Peripheral quantitative computed tomography of the radius showed a decrease in trabecular volumetric BMD at the distal metaphysis of 19% but an increase in cortical volumetric BMD of 4% (p\u2009&amp;lt;\u20090.05 for both). None of the patients sustained a new vertebral compression fracture during follow-up. The proportion of patients with new long-bone fractures was higher in the 2\u2009years before treatment discontinuation than in the last 2\u2009years of follow-up (42% and 16%, respectively; p\u2009&amp;lt;\u20090.05). \u00a9 2019 American Society for Bone and Mineral Research.<\/jats:p>","DOI":"10.1002\/jbmr.3833","type":"journal-article","created":{"date-parts":[[2019,7,29]],"date-time":"2019-07-29T18:33:40Z","timestamp":1564425220000},"page":"2198-2204","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":17,"title":["Osteogenesis Imperfecta: Skeletal Outcomes After Bisphosphonate Discontinuation at Final Height"],"prefix":"10.1093","volume":"34","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2539-227X","authenticated-orcid":false,"given":"Marie-Eve","family":"Robinson","sequence":"first","affiliation":[{"name":"Shriners Hospital for Children Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pamela","family":"Trejo","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Telma","family":"Palomo","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6616-3484","authenticated-orcid":false,"given":"Francis H","family":"Glorieux","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6261-0360","authenticated-orcid":false,"given":"Frank","family":"Rauch","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2019,7,29]]},"reference":[{"key":"2024021014415400800_jbmr3833-bib-0001","doi-asserted-by":"crossref","first-page":"1657","DOI":"10.1016\/S0140-6736(15)00728-X","article-title":"Osteogenesis imperfecta","volume":"387","author":"Forlino","year":"2016","journal-title":"Lancet"},{"key":"2024021014415400800_jbmr3833-bib-0002","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"},{"key":"2024021014415400800_jbmr3833-bib-0003","doi-asserted-by":"crossref","DOI":"10.1002\/jbm4.10174","article-title":"Osteogenesis imperfecta: new perspectives from clinical and translational research","author":"Tauer","year":"2019","journal-title":"JBMR Plus."},{"key":"2024021014415400800_jbmr3833-bib-0004","doi-asserted-by":"crossref","first-page":"158","DOI":"10.1186\/s13023-018-0905-4","article-title":"Consensus statement on physical rehabilitation in children and adolescents with osteogenesis imperfecta","volume":"13","author":"Mueller","year":"2018","journal-title":"Orphanet J Rare Dis."},{"key":"2024021014415400800_jbmr3833-bib-0005","doi-asserted-by":"crossref","first-page":"2150","DOI":"10.1002\/jbmr.2567","article-title":"Intravenous bisphosphonate therapy of young children with osteogenesis imperfecta: skeletal findings during follow up throughout the growing years","volume":"30","author":"Palomo","year":"2015","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0006","doi-asserted-by":"crossref","first-page":"321","DOI":"10.1159\/000331128","article-title":"Reshaping of vertebrae during treatment with neridronate or pamidronate in children with osteogenesis imperfecta","volume":"76","author":"Semler","year":"2011","journal-title":"Horm Res Paediatr."},{"key":"2024021014415400800_jbmr3833-bib-0007","doi-asserted-by":"crossref","first-page":"3427","DOI":"10.1007\/s00198-016-3723-3","article-title":"Osteogenesis imperfecta in children and adolescents\u2013new developments in diagnosis and treatment","volume":"27","author":"Trejo","year":"2016","journal-title":"Osteoporos Int."},{"key":"2024021014415400800_jbmr3833-bib-0008","doi-asserted-by":"crossref","first-page":"120","DOI":"10.1007\/s00223-010-9383-y","article-title":"Bone mineral density and fracture rate in response to intravenous and oral bisphosphonates in adult osteogenesis imperfecta","volume":"87","author":"Shapiro","year":"2010","journal-title":"Calcif Tissue Int."},{"key":"2024021014415400800_jbmr3833-bib-0009","doi-asserted-by":"crossref","first-page":"1377","DOI":"10.1016\/S0140-6736(04)16051-0","article-title":"Osteogenesis imperfecta","volume":"363","author":"Rauch","year":"2004","journal-title":"Lancet."},{"key":"2024021014415400800_jbmr3833-bib-0010","doi-asserted-by":"crossref","first-page":"45","DOI":"10.1542\/peds.109.1.45","article-title":"Centers for Disease Control and Prevention 2000 growth charts for the United States: improvements to the 1977 National Center for Health Statistics version","volume":"109","author":"Ogden","year":"2002","journal-title":"Pediatrics."},{"key":"2024021014415400800_jbmr3833-bib-0011","doi-asserted-by":"crossref","first-page":"206","DOI":"10.1002\/jbmr.1730","article-title":"Bone mineral content and density of the lumbar spine of infants and toddlers: influence of age, sex, race, growth, and human milk feeding","volume":"28","author":"Kalkwarf","year":"2013","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0012","doi-asserted-by":"crossref","first-page":"3160","DOI":"10.1210\/jc.2011-1111","article-title":"Revised reference curves for bone mineral content and areal bone mineral density according to age and sex for black and non-black children: results of the bone mineral density in childhood study","volume":"96","author":"Zemel","year":"2011","journal-title":"J Clin Endocrinol Metab."},{"key":"2024021014415400800_jbmr3833-bib-0013","first-page":"217","article-title":"Peripheral quantitative computed tomography of the proximal radius in young subjects\u2014new reference data and interpretation of results","volume":"8","author":"Rauch","year":"2008","journal-title":"J Musculoskelet Neuronal Interact."},{"key":"2024021014415400800_jbmr3833-bib-0014","first-page":"119","article-title":"Peripheral quantitative computed tomography of the distal radius in young subjects\u2014new reference data and interpretation of results","volume":"5","author":"Rauch","year":"2005","journal-title":"J Musculoskelet Neuronal Interact."},{"key":"2024021014415400800_jbmr3833-bib-0015","doi-asserted-by":"crossref","first-page":"1137","DOI":"10.1002\/jbmr.5650080915","article-title":"Vertebral fracture assessment using a semiquantitative technique","volume":"8","author":"Genant","year":"1993","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0016","doi-asserted-by":"crossref","first-page":"2129","DOI":"10.1007\/s00198-011-1819-3","article-title":"Agreement between semi-automatic radiographic morphometry and Genant semi-quantitative method in the assessment of vertebral fractures","volume":"23","author":"Sanfelix-Genoves","year":"2012","journal-title":"Osteoporos Int."},{"key":"2024021014415400800_jbmr3833-bib-0017","doi-asserted-by":"crossref","first-page":"1547","DOI":"10.1007\/s00198-008-0832-7","article-title":"Spinal deformity index (SDI) is a good predictor of incident vertebral fractures","volume":"20","author":"Kerkeni","year":"2009","journal-title":"Osteoporos Int."},{"key":"2024021014415400800_jbmr3833-bib-0018","doi-asserted-by":"crossref","first-page":"1191","DOI":"10.1359\/JBMR.040303","article-title":"Sclerotic metaphyseal lines in a child treated with pamidronate: histomorphometric analysis","volume":"19","author":"Rauch","year":"2004","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0019","doi-asserted-by":"crossref","first-page":"374","DOI":"10.1359\/JBMR.051207","article-title":"Cyclical intravenous pamidronate treatment affects metaphyseal modeling in growing patients with osteogenesis imperfecta","volume":"21","author":"Land","year":"2006","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0020","doi-asserted-by":"crossref","first-page":"1405","DOI":"10.1002\/jbmr.1592","article-title":"Bone geometry, density, and microarchitecture in the distal radius and tibia in adults with osteogenesis imperfecta type I assessed by high-resolution pQCT","volume":"27","author":"Folkestad","year":"2012","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0021","doi-asserted-by":"crossref","first-page":"125","DOI":"10.1002\/jbmr.2920","article-title":"Fracture rates and fracture sites in patients with osteogenesis imperfecta: a nationwide register-based cohort study","volume":"32","author":"Folkestad","year":"2017","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0022","doi-asserted-by":"crossref","first-page":"821","DOI":"10.1016\/j.bone.2006.11.020","article-title":"Long-bone changes after pamidronate discontinuation in children and adolescents with osteogenesis imperfecta","volume":"40","author":"Rauch","year":"2007","journal-title":"Bone."},{"key":"2024021014415400800_jbmr3833-bib-0023","doi-asserted-by":"crossref","first-page":"63","DOI":"10.1515\/JPEM.2008.21.1.63","article-title":"Cyclic pamidronate therapy in children with osteogenesis imperfecta: results of treatment and follow-up after discontinuation","volume":"21","author":"Andiran","year":"2008","journal-title":"J Pediatr Endocrinol Metab."},{"key":"2024021014415400800_jbmr3833-bib-0024","doi-asserted-by":"crossref","first-page":"243","DOI":"10.1002\/jbmr.1494","article-title":"The effect of 3 versus 6 years of zoledronic acid treatment of osteoporosis: a randomized extension to the HORIZON-Pivotal Fracture Trial (PFT)","volume":"27","author":"Black","year":"2012","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0025","doi-asserted-by":"crossref","first-page":"934","DOI":"10.1002\/jbmr.2442","article-title":"The effect of 6 versus 9 years of zoledronic acid treatment in osteoporosis: a randomized second extension to the HORIZON-Pivotal Fracture Trial (PFT)","volume":"30","author":"Black","year":"2015","journal-title":"J Bone Miner Res."},{"key":"2024021014415400800_jbmr3833-bib-0026","doi-asserted-by":"crossref","first-page":"190","DOI":"10.1002\/jbmr.3337","article-title":"Vertebral fractures after discontinuation of denosumab: a post hoc analysis of the randomized placebo-controlled FREEDOM trial and its extension","volume":"33","author":"Cummings","year":"2018","journal-title":"J Bone Miner Res."}],"container-title":["Journal of Bone and Mineral Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/jbmr.3833","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/jbmr.3833","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/academic.oup.com\/jbmr\/article-pdf\/34\/12\/2198\/56654601\/jbmr3833.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jbmr\/article-pdf\/34\/12\/2198\/56654601\/jbmr3833.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,3,13]],"date-time":"2024-03-13T15:04:38Z","timestamp":1710342278000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jbmr\/article\/34\/12\/2198-2204\/7380894"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,7,29]]},"references-count":26,"journal-issue":{"issue":"12","published-online":{"date-parts":[[2019,7,29]]},"published-print":{"date-parts":[[2019,12,1]]}},"URL":"https:\/\/doi.org\/10.1002\/jbmr.3833","relation":{},"ISSN":["0884-0431","1523-4681"],"issn-type":[{"value":"0884-0431","type":"print"},{"value":"1523-4681","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2019,12,1]]},"published":{"date-parts":[[2019,7,29]]},"assertion":[{"value":"2019-05-11","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2019-07-18","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2019-08-26","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}