{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,21]],"date-time":"2026-10-21T16:50:26Z","timestamp":1792601426418,"version":"4.5.0"},"reference-count":43,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2020,6,26]],"date-time":"2020-06-26T00:00:00Z","timestamp":1593129600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Orthopaedic Surgery"],"published-print":{"date-parts":[[2020,8]]},"abstract":"<jats:sec><jats:title>Objective<\/jats:title><jats:p>To assess the effectiveness and safety of oral bisphosphonates in increasing bone mineral density (BMD), reducing fractures, and improving clinical function in patients with osteogenesis imperfecta (OI).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Studies were eligible for inclusion if they were randomized controlled trials of directly comparing oral bisphosphonate therapy with placebo\u2010group in OI patients. Data synthesis regarding to bone mineral density as measured by dual\u2010energy X\u2010ray absorptiometry (DEXA), decreased fracture incidence, change in biochemical markers of bone and mineral metabolism, bone histology, growth, bone pain, quality of life, and others were assessed, and meta\u2010analysis done when possible.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>From 98 potential references and six randomized controlled studies a total of 263 participants receiving oral bisphosphonates and 143 placebo treatments contributed data to meta\u2010analysis. Pooled meta\u2010analysis of three studies suggested that there was significant difference between bisphosphonate treated group and placebo in number of patients with at least one fracture (mean difference 0.53, 95% confidence interval 0.32\u20130.89, <jats:italic>P<\/jats:italic> = 0.02). Pooled meta\u2010analysis of two studies suggested that significant difference was noted between bisphosphonate treated group and placebo in mean percentage change in spine BMD (T\u2010score) (mean difference 28.43, 95% confidence interval 7.09\u201049.77, <jats:italic>P<\/jats:italic> = 0.009). The similar effect was shown in the term of mean change (Z\u2010score) in spine BMD.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Significant improvement in lumbar areal BMD in patients affected with OI has been shown when treated with oral bisphosphonates, even though only a small population was enrolled. We cannot draw a definite conclusion that the increase in BMD can be translated into fracture reduction and clinical functional improvement. The optimal method, dose, type, initiation, and duration of oral bisphosphonates therapy still remains unclear. Well\u2010designed, adequately\u2010powered, placebo\u2010controlled RCTs investigating the effects of oral bisphosphonates on fractures reduction and improvement in quality of life in both children and adults are studied here.<\/jats:p><\/jats:sec>","DOI":"10.1111\/os.12611","type":"journal-article","created":{"date-parts":[[2020,6,27]],"date-time":"2020-06-27T08:58:35Z","timestamp":1593248315000},"page":"1293-1303","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta\u2010Analysis of Six Randomized Placebo\u2010Controlled Trials"],"prefix":"10.1111","volume":"12","author":[{"given":"Zhi\u2010min","family":"Ying","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery, the Second Affiliated Hospital School of Medicine, Zhejiang University  Zhejiang China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bin","family":"Hu","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, the Second Affiliated Hospital School of Medicine, Zhejiang University  Zhejiang China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1676-8474","authenticated-orcid":false,"given":"Shi\u2010gui","family":"Yan","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, the Second Affiliated Hospital School of Medicine, Zhejiang University  Zhejiang China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,6,26]]},"reference":[{"key":"e_1_2_5_2_1","first-page":"397","article-title":"Osteogenesis imperfecta","volume":"10","author":"Ben Amor M","year":"2013","journal-title":"Pediatr Endocrinol Rev"},{"key":"e_1_2_5_3_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2013-1505"},{"key":"e_1_2_5_4_1","unstructured":"SteinerRD PepinMG ByersPH.Gene Reviews; Osteogenesis ImperfectaAvailable from:www.genetests.org(accessed 28 January 2005)."},{"key":"e_1_2_5_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.16.2.101"},{"key":"e_1_2_5_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(04)16051-0"},{"key":"e_1_2_5_7_1","first-page":"293","article-title":"Consistent linkage of dominantly inherited osteogenesis: imperfecta to the type I collagen loci: COL 1A1 and COL 1A2","volume":"46","author":"Sykes B","year":"1990","journal-title":"Am J Hum Genet"},{"key":"e_1_2_5_8_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.28.7.433"},{"key":"e_1_2_5_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.1989.tb03198.x"},{"key":"e_1_2_5_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/s004310051322"},{"key":"e_1_2_5_11_1","doi-asserted-by":"publisher","DOI":"10.1177\/000348940211101014"},{"key":"e_1_2_5_12_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMct1004903"},{"key":"e_1_2_5_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1998.tb01388.x"},{"key":"e_1_2_5_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(97)70074-X"},{"key":"e_1_2_5_15_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199810013391402"},{"key":"e_1_2_5_16_1","doi-asserted-by":"publisher","DOI":"10.1136\/adc.2006.096552"},{"key":"e_1_2_5_17_1","first-page":"1846","article-title":"Pamidronate treatment of severe osteogenesis imperfecta in children under 3\u2009years of age","volume":"85","author":"Plotkin H","year":"2000","journal-title":"J Clin Endocrinol Metab"},{"key":"e_1_2_5_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2004.07.003"},{"key":"e_1_2_5_19_1","doi-asserted-by":"publisher","DOI":"10.1136\/adc.2007.125468"},{"key":"e_1_2_5_20_1","doi-asserted-by":"publisher","DOI":"10.1359\/jbmr.2003.18.1.126"},{"key":"e_1_2_5_21_1","doi-asserted-by":"publisher","DOI":"10.1359\/JBMR.041232"},{"key":"e_1_2_5_22_1","doi-asserted-by":"publisher","DOI":"10.1359\/JBMR.050109"},{"key":"e_1_2_5_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00431-006-0399-2"},{"key":"e_1_2_5_24_1","doi-asserted-by":"publisher","DOI":"10.5435\/00124635-200507000-00002"},{"key":"e_1_2_5_25_1","doi-asserted-by":"publisher","DOI":"10.1097\/BLO.0b013e31802c9098"},{"key":"e_1_2_5_26_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.323.7303.42"},{"key":"e_1_2_5_27_1","unstructured":"HigginsJPT GreenS Cochrane handbook for systematic reviews of interventions version 5.1.0. 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