{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,12]],"date-time":"2026-11-12T18:32:43Z","timestamp":1794508363738,"version":"4.9.1"},"reference-count":50,"publisher":"Oxford University Press (OUP)","issue":"9","license":[{"start":{"date-parts":[[2024,7,10]],"date-time":"2024-07-10T00:00:00Z","timestamp":1720569600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"DOI":"10.13039\/100011781","name":"Shriners Hospitals for Children","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100011781","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000156","name":"Fonds de Recherche du Qu\u00e9bec - Sant\u00e9","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000156","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,9,2]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Osteogenesis imperfecta (OI) is a skeletal dysplasia characterized by low bone mass and frequent fractures. Children with OI are commonly treated with bisphosphonates to reduce fracture rate, but treatment options for adults are limited. In the Phase 2b ASTEROID trial, setrusumab (a sclerostin neutralizing antibody, SclAb) improved bone density and strength in adults with type I, III, and IV OI. Here, we investigate bone matrix material properties in tetracycline-labeled trans iliac biopsies from 3 groups: (1) control: individuals with no metabolic bone disease, (2) OI: individuals with OI, (3) SclAb-OI: individuals with OI after 6 mo of setrusumab treatment (as part of the ASTEROID trial). In addition to bone histomorphometry, bone mineral and matrix properties were evaluated with nanoindentation, Raman spectroscopy, second harmonic generation imaging, quantitative backscatter electron imaging, and small-angle X-ray scattering. Spatial locations of fluorochrome labels were identified to differentiate inter-label bone of the same tissue age and intra-cortical bone. No difference in collagen orientation was found between the groups. The bone mineral density distribution and analysis of Raman spectra indicate that OI groups have greater mean mineralization, greater relative mineral content, and lower crystallinity than the control group, which was not altered by SclAb treatment. Finally, a lower modulus and hardness were measured in the inter-label bone of the OI-SclAb group compared to the OI group. Previous studies suggest that even though bone from OI has a higher mineral content, the extracellular matrix (ECM) has comparable mechanical properties. Therefore, fragility in OI may stem from contributions from other yet unexplored aspects of bone organization at higher length scales. We conclude that SclAb treatment leads to increased bone mass while not adversely affecting bone matrix properties in individuals with OI.<\/jats:p>","DOI":"10.1093\/jbmr\/zjae108","type":"journal-article","created":{"date-parts":[[2024,7,10]],"date-time":"2024-07-10T06:16:04Z","timestamp":1720592164000},"page":"1229-1239","source":"Crossref","is-referenced-by-count":9,"title":["Bone matrix properties in adults with osteogenesis imperfecta are not adversely affected by setrusumab\u2014a sclerostin neutralizing antibody"],"prefix":"10.1093","volume":"39","author":[{"given":"Maximilian","family":"Rummler","sequence":"first","affiliation":[{"name":"Research Centre, Shriners Hospital for Children , Montreal, QC H4A 0A9, Canada"},{"name":"Department of Experimental Surgery, McGill University , Montreal, QC H3A 0G4, Canada"},{"name":"Department of Biomaterials, Max Planck Institute of Colloids and Interfaces , Potsdam 14476, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Victoria","family":"Schemenz","sequence":"additional","affiliation":[{"name":"Department of Biomaterials, Max Planck Institute of Colloids and Interfaces , Potsdam 14476, Germany"},{"name":"Department for Operative , Preventive and Pediatric Dentistry, Centrum f\u00fcr Zahn-, Mund- und Kieferheilkunde, , Berlin 14197, Germany"},{"name":"Charit\u00e9 - Universit\u00e4tsmedizin , Preventive and Pediatric Dentistry, Centrum f\u00fcr Zahn-, Mund- und Kieferheilkunde, , Berlin 14197, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Samantha","family":"McCluskey","sequence":"additional","affiliation":[{"name":"Research Centre, Shriners Hospital for Children , Montreal, QC H4A 0A9, Canada"},{"name":"Faculty of Dental Medicine and Oral Health Sciences, McGill University , Montreal, QC H3A 0G4, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anton","family":"Davydok","sequence":"additional","affiliation":[{"name":"Institute of Material Physics , Helmholtz Zentrum Hereon, Hamburg 22607, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank","family":"Rauch","sequence":"additional","affiliation":[{"name":"Research Centre, Shriners Hospital for Children , Montreal, QC H4A 0A9, Canada"},{"name":"Department of Pediatrics, McGill University , Montreal, QC H3A 0G4, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francis H","family":"Glorieux","sequence":"additional","affiliation":[{"name":"Research Centre, Shriners Hospital for Children , Montreal, QC H4A 0A9, Canada"},{"name":"Department of Pediatrics, McGill University , Montreal, QC H3A 0G4, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew J","family":"Harrington","sequence":"additional","affiliation":[{"name":"Department of Chemistry, McGill University , Montreal, QC, H3A 0G4, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wolfgang","family":"Wagermaier","sequence":"additional","affiliation":[{"name":"Department of Biomaterials, Max Planck Institute of Colloids and Interfaces , Potsdam 14476, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2907-3580","authenticated-orcid":false,"given":"Bettina M","family":"Willie","sequence":"additional","affiliation":[{"name":"Research Centre , Shriners Hospital for Children, Montreal, QC H4A 0A9, Canada"},{"name":"Department of Experimental Surgery, McGill University , Montreal, QC H3A 0G4, Canada"},{"name":"Faculty of Dental Medicine and Oral Health Sciences, McGill University , Montreal, QC H3A 0G4, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9927-3372","authenticated-orcid":false,"given":"Elizabeth A","family":"Zimmermann","sequence":"additional","affiliation":[{"name":"Research Centre, Shriners Hospital for Children , Montreal, QC H4A 0A9, Canada"},{"name":"Faculty of Dental Medicine and Oral Health Sciences, McGill University , Montreal, QC H3A 0G4, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2024,7,10]]},"reference":[{"key":"2024090404570905300_ref1","doi-asserted-by":"crossref","first-page":"871","DOI":"10.1002\/9781119266594.ch113","volume-title":"Primer on the Metabolic Bone Diseases and Disorders of Mineral Metabolism","author":"Marini","year":"2018"},{"issue":"9418","key":"2024090404570905300_ref2","doi-asserted-by":"crossref","first-page":"1377","DOI":"10.1016\/S0140-6736(04)16051-0","article-title":"Osteogenesis 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