{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T21:22:54Z","timestamp":1791926574426,"version":"4.3.4"},"reference-count":24,"publisher":"American Academy of Pediatrics (AAP)","issue":"3","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2003,3,1]]},"abstract":"<jats:p>Objective. Several studies have reported beneficial effects of bisphosphonates in children with osteogenesis imperfecta (OI); however, these studies have differed in the protocols they used, and none has been independently replicated. We intended to confirm the efficacy of a specific intravenous bisphosphonate protocol in children with moderate to severe OI.<\/jats:p>\n               <jats:p>Methods. We used the protocol described by Glorieux et al and performed a prospective clinical trial in 6 children who were aged 22 months to 14 years. Each patient received intravenous pamidronate therapy for a minimum of 2 years in cycles of 1 mg\/kg daily over 3 consecutive days at a mean cycle interval of 3.8 months. Outcome measures included lumbar spine areal bone mineral density (BMD) and z score, fracture rate, and occupational therapy functional assessment with serial Pediatric Evaluation of Disability Inventory.<\/jats:p>\n               <jats:p>Results. While on therapy, the average annual increase in areal BMD was 48% and the average annual increase in BMD z score was 1.0. This increase in z score is statistically significant. There was no clear correlation between changes in BMD and fracture rate. All patients experienced functional improvement in mobility.<\/jats:p>\n               <jats:p>Conclusions. Our results support the findings of Glorieux et al that cyclic administration of intravenous pamidronate in children with OI has beneficial effects with respect to BMD z scores and physical disability. Long-term follow-up will be required to determine whether bisphosphonate therapy will decrease fracture rates and increase mobility in children with moderate to severe OI.<\/jats:p>","DOI":"10.1542\/peds.111.3.573","type":"journal-article","created":{"date-parts":[[2004,8,14]],"date-time":"2004-08-14T00:43:47Z","timestamp":1092444227000},"page":"573-578","source":"Crossref","is-referenced-by-count":82,"title":["Intravenous Bisphosphonate Therapy in Children With Osteogenesis Imperfecta"],"prefix":"10.1542","volume":"111","author":[{"given":"Marni J.","family":"Falk","sequence":"first","affiliation":[{"name":"Department of Genetics and Center for Human Genetics"},{"name":"Department of Pediatrics"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shauna","family":"Heeger","sequence":"additional","affiliation":[{"name":"Department of Genetics and Center for Human Genetics"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katherine A.","family":"Lynch","sequence":"additional","affiliation":[{"name":"Department of Genetics and Center for Human Genetics"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathleen R.","family":"DeCaro","sequence":"additional","affiliation":[{"name":"General Clinical Research Center"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Deborah","family":"Bohach","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karen S.","family":"Gibson","sequence":"additional","affiliation":[{"name":"University Suburban Health Center, University Hospitals of Cleveland and Case Western Reserve University School of Medicine, Cleveland, Ohio"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew L.","family":"Warman","sequence":"additional","affiliation":[{"name":"Department of Genetics and Center for Human Genetics"},{"name":"Department of Pediatrics"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"417","published-online":{"date-parts":[[2003,3,1]]},"reference":[{"key":"2021120919021534300_R1","unstructured":"Marini JC, Chernoff EJ. 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