{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,25]],"date-time":"2026-10-25T02:59:44Z","timestamp":1792897184259,"version":"4.5.0"},"reference-count":82,"publisher":"Oxford University Press (OUP)","issue":"11","license":[{"start":{"date-parts":[[2024,8,21]],"date-time":"2024-08-21T00:00:00Z","timestamp":1724198400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000038","name":"Natural Sciences and Engineering Research Council","doi-asserted-by":"crossref","award":["RGPIN-288253"],"award-info":[{"award-number":["RGPIN-288253"]}],"id":[{"id":"10.13039\/501100000038","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100000024","name":"Canadian Institutes for Health Research","doi-asserted-by":"crossref","award":["CIHR PJT-74421"],"award-info":[{"award-number":["CIHR PJT-74421"]}],"id":[{"id":"10.13039\/501100000024","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Shriners of North America Developmental","award":["DGBR 87210-CAN-24"],"award-info":[{"award-number":["DGBR 87210-CAN-24"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,10,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Osteogenesis imperfecta (OI) is a rare bone fragility disorder caused by mutations in genes encoding collagen type I or that affect its processing. Alterations in osteoclasts were suggested to contribute to OI pathophysiology. We aimed to systematically identify studies reporting measures of osteoclast formation and function in patients and mouse models of OI, to quantify OI-induced changes. The systematic search of Medline, Ovid, and Web of Science identified 798 unique studies. After screening, we included 23 studies for meta-analysis, reporting osteoclast parameters in 310 patients with OI of 9 different types and 16 studies reporting osteoclast parameters in 406 animals of 11 different OI mouse models. The standardized mean difference with 95% confidence interval (CI) was used as the effect size, and random-effects meta-analysis was performed. In patients with OI, collagen degradation markers were significantly higher compared with age-matched controls, with an effect size of 1.23 (CI: 0.36, 2.10]. Collagen degradation markers were the most elevated in the 3- to 7-year-old age group and in patients with more severe forms of OI. Bone histomorphometry demonstrated the trends for higher osteoclast numbers (1.16; CI: \u22120.22, 2.55) and osteoclast surface (0.43; CI: \u22120.63, 1.49), and significantly higher eroded surface (3.24; CI: 0.51, 5.96) compared with age-matched controls. In OI mice, meta-analysis demonstrated significant increases in collagen degradation markers (1.59; CI: 1.07, 2.11), in osteoclast numbers (0.94; CI: 0.50, 1.39), osteoclast surface (0.73; CI: 0.22, 1.23), and eroded surface (1.31; CI: 0.54, 2.08). The largest differences were in OI mice with the mutations in Col1a1 and Col1a2 genes. There were no differences between males and females in clinical or animal studies. Quantitative estimates of changes in osteoclast indices and their variance for patients with OI are important for planning future studies. We confirmed that similar changes are observed in mice with OI, supporting their translational utility.<\/jats:p>","DOI":"10.1093\/jbmrpl\/ziae112","type":"journal-article","created":{"date-parts":[[2024,8,22]],"date-time":"2024-08-22T21:26:47Z","timestamp":1724362007000},"source":"Crossref","is-referenced-by-count":12,"title":["Osteoclast indices in osteogenesis imperfecta: systematic review and meta-analysis"],"prefix":"10.1093","volume":"8","author":[{"given":"Sirion","family":"Aksornthong","sequence":"first","affiliation":[{"name":"Department of Experimental Surgery, McGill University , Montreal, Quebec H3G 1A4,","place":["Canada"]},{"name":"Shriners Hospital for Children\u2014Canada , Montreal, Quebec H4A 0A9,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Priyesh","family":"Patel","sequence":"additional","affiliation":[{"name":"Shriners Hospital for Children\u2014Canada , Montreal, Quebec H4A 0A9,","place":["Canada"]},{"name":"Faculty of Dental Medicine and Oral Health Sciences, McGill University , Montreal, Quebec H3A 1G1,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3570-3147","authenticated-orcid":false,"given":"Svetlana V","family":"Komarova","sequence":"additional","affiliation":[{"name":"Department of Experimental Surgery, McGill University , Montreal, Quebec H3G 1A4,","place":["Canada"]},{"name":"Shriners Hospital for Children\u2014Canada , Montreal, Quebec H4A 0A9,","place":["Canada"]},{"name":"Faculty of Dental Medicine and Oral Health Sciences, McGill University , Montreal, Quebec H3A 1G1,","place":["Canada"]},{"name":"Department of Biomedical Engineering , Faculty of Engineering, , Edmonton, Alberta T6G 1H9,","place":["Canada"]},{"name":"University of Alberta , Faculty of Engineering, , Edmonton, Alberta T6G 1H9,","place":["Canada"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2024,8,21]]},"reference":[{"issue":"9418","key":"2024100408410000100_ref1","doi-asserted-by":"publisher","first-page":"1377","DOI":"10.1016\/s0140-6736(04)16051-0","article-title":"Osteogenesis imperfecta","volume":"363","author":"Rauch","year":"2004","journal-title":"Lancet"},{"issue":"6","key":"2024100408410000100_ref2","doi-asserted-by":"publisher","first-page":"381","DOI":"10.1097\/med.0000000000000367","article-title":"Osteogenesis imperfecta: diagnosis and treatment","volume":"24","author":"Palomo","year":"2017","journal-title":"Curr Opin Endocrinol Diabetes Obes"},{"issue":"6","key":"2024100408410000100_ref3","doi-asserted-by":"publisher","first-page":"708","DOI":"10.1097\/MOP.0000000000000813","article-title":"Osteogenesis imperfecta: advancements in genetics and treatment","volume":"31","author":"Rossi","year":"2019","journal-title":"Curr Opin 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