{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,2]],"date-time":"2026-07-02T03:51:29Z","timestamp":1782964289761,"version":"3.54.5"},"reference-count":38,"publisher":"Oxford University Press (OUP)","issue":"12","license":[{"start":{"date-parts":[[2015,6,8]],"date-time":"2015-06-08T00:00:00Z","timestamp":1433721600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"name":"This work was supported by grants from the Chercheur-Boursier Clinicien program of the Fonds de Recherche du Qu\u00e9bec - Sant\u00e9 (to FR); and the Shriners of North America."}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2015,12,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:sec>\n                  <jats:title>\u2003<\/jats:title>\n                  <jats:p>Cyclical intravenous bisphosphonate therapy is widely used to treat children with osteogenesis imperfecta (OI), but little is known about long-term treatment outcomes. We therefore reviewed 37 children with OI (OI type I, n\u2009=\u20091; OI type III, n\u2009=\u200914; and OI type IV, n\u2009=\u200922) who started intravenous bisphosphonate therapy before 5 years of age (median 2.2 years; range, 0.1 to 4.8 years), and who had a subsequent follow-up period of at least 10 years (median 14.8 years; range, 10.7 to 18.2 years), during which they had received intravenous bisphosphonate treatment (pamidronate or zoledronic acid) for at least 6 years. During the observation period, the mean lumbar spine areal bone mineral density Z-score increased from \u20136.6 (SD 3.1) to \u20133.0 (SD 1.8), and weight Z-score increased from \u20132.3 (SD 1.5) to \u20131.7 (SD 1.7) (p\u2009&amp;lt;\u20090.001 and p\u2009=\u20090.008). At the time of the last assessment, patients with OI type IV had significantly higher height Z-scores than a control group of patients matched for age, gender, and OI type who had not received bisphosphonates. Patients had a median of six femur fractures (range, 0 to 18) and five tibia fractures (range, 0 to 17) during the follow-up period. At baseline, 35% of vertebra were affected by compression fractures, whereas only 6% of vertebra appeared compressed at the last evaluation (p\u2009&amp;lt;\u20090.001), indicating vertebral reshaping during growth. Spinal fusion surgery was performed in 16 patients (43%). Among the 21 patients who did not have spinal fusion surgery, 13 had scoliosis with a curvature ranging from 10 to 56 degrees. In conclusion, long-term intravenous bisphosphonate therapy was associated with higher Z-scores for lumbar spine areal bone mineral density and vertebral reshaping, but long-bone fracture rates were still high and the majority of patients developed scoliosis. \u00a9 2015 American Society for Bone and Mineral Research.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1002\/jbmr.2567","type":"journal-article","created":{"date-parts":[[2015,6,9]],"date-time":"2015-06-09T16:51:44Z","timestamp":1433868704000},"page":"2150-2157","source":"Crossref","is-referenced-by-count":126,"title":["Intravenous Bisphosphonate Therapy of Young Children With Osteogenesis Imperfecta: Skeletal Findings During Follow Up Throughout the Growing Years"],"prefix":"10.1093","volume":"30","author":[{"given":"Telma","family":"Palomo","sequence":"first","affiliation":[{"name":"Shriners Hospital for Children and McGill UniversityMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fran\u00e7ois","family":"Fassier","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children and McGill UniversityMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean","family":"Ouellet","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children and McGill UniversityMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Atsuko","family":"Sato","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children and McGill UniversityMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathleen","family":"Montpetit","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children and McGill UniversityMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francis H","family":"Glorieux","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children and McGill UniversityMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank","family":"Rauch","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children and McGill UniversityMontrealQuebecCanada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2015,6,8]]},"reference":[{"key":"2024020318595409400_jbmr2567-bib-0001","doi-asserted-by":"crossref","first-page":"540","DOI":"10.1038\/nrendo.2011.81","article-title":"New perspectives on osteogenesis imperfecta","volume":"7","author":"Forlino","year":"2011","journal-title":"Nat Rev Endocrinol."},{"key":"2024020318595409400_jbmr2567-bib-0002","doi-asserted-by":"crossref","first-page":"1377","DOI":"10.1016\/S0140-6736(04)16051-0","article-title":"Osteogenesis 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