{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,11]],"date-time":"2026-09-11T10:31:47Z","timestamp":1789122707462,"version":"build-2803163510"},"reference-count":106,"publisher":"Elsevier BV","license":[{"start":{"date-parts":[[2024,7,1]],"date-time":"2024-07-01T00:00:00Z","timestamp":1719792000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2024,7,1]],"date-time":"2024-07-01T00:00:00Z","timestamp":1719792000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2024,5,11]],"date-time":"2024-05-11T00:00:00Z","timestamp":1715385600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100002920","name":"Research Grants Council, University Grants Committee","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100002920","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001747","name":"Hong Kong Baptist University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001747","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100012166","name":"National Key Research and Development Program of China","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100012166","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100016104","name":"Key Project of Research and Development Plan of Hunan Province","doi-asserted-by":"publisher","award":["2022WK2010"],"award-info":[{"award-number":["2022WK2010"]}],"id":[{"id":"10.13039\/100016104","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002855","name":"Ministry of Science and Technology of the People&apos;s Republic of China","doi-asserted-by":"publisher","award":["2018YFA0800804"],"award-info":[{"award-number":["2018YFA0800804"]}],"id":[{"id":"10.13039\/501100002855","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["82300988"],"award-info":[{"award-number":["82300988"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["81974123"],"award-info":[{"award-number":["81974123"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100007162","name":"Guangdong Provincial Department of Science and Technology","doi-asserted-by":"publisher","award":["2019B1515120089"],"award-info":[{"award-number":["2019B1515120089"]}],"id":[{"id":"10.13039\/501100007162","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Journal of Orthopaedic Translation"],"published-print":{"date-parts":[[2024,7]]},"DOI":"10.1016\/j.jot.2024.05.004","type":"journal-article","created":{"date-parts":[[2024,6,19]],"date-time":"2024-06-19T13:24:54Z","timestamp":1718803494000},"page":"39-49","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":9,"special_numbering":"C","title":["Sclerostin inhibition in rare bone diseases: Molecular understanding and therapeutic perspectives"],"prefix":"10.1016","volume":"47","author":[{"given":"Tao","family":"Xiaohui","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Luyao","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1667-5777","authenticated-orcid":false,"given":"Xin","family":"Yang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hewen","family":"Jiang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ning","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Huarui","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dijie","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaofei","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yihao","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shenghang","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chuanxin","family":"Zhong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sifan","family":"Yu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Meishen","family":"Ren","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Meiheng","family":"Sun","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nanxi","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tienan","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuan","family":"Ma","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fangfei","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jin","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuanyuan","family":"Yu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hua","family":"Yue","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhenlin","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7807-7695","authenticated-orcid":false,"given":"Ge","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"7","key":"10.1016\/j.jot.2024.05.004_bib1","doi-asserted-by":"crossref","first-page":"E1526","DOI":"10.1002\/humu.21274","article-title":"First missense mutation in the SOST gene causing sclerosteosis by loss of sclerostin function","volume":"31","author":"Piters","year":"2010","journal-title":"Hum Mutat"},{"issue":"6","key":"10.1016\/j.jot.2024.05.004_bib2","doi-asserted-by":"crossref","first-page":"1661","DOI":"10.1086\/302416","article-title":"Localization of the gene for sclerosteosis to the van Buchem disease-gene region on chromosome 17q12-q21","volume":"64","author":"Balemans","year":"1999","journal-title":"Am J Hum Genet"},{"issue":"5","key":"10.1016\/j.jot.2024.05.004_bib3","doi-asserted-by":"crossref","first-page":"1035","DOI":"10.1002\/jbmr.304","article-title":"PTH receptor signaling in osteocytes governs periosteal bone formation and intracortical remodeling","volume":"26","author":"Rhee","year":"2011","journal-title":"J Bone Miner Res"},{"key":"10.1016\/j.jot.2024.05.004_bib4","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.jot.2015.08.004","article-title":"Sclerostin, an emerging therapeutic target for treating osteoporosis and osteoporotic fracture: a general review","volume":"4","author":"Suen","year":"2016","journal-title":"J Orthop Translat"},{"issue":"4","key":"10.1016\/j.jot.2024.05.004_bib5","doi-asserted-by":"crossref","first-page":"R95","DOI":"10.1530\/EJE-20-0299","article-title":"Osteogenesis imperfecta: an update on clinical features and therapies","volume":"183","author":"Marom","year":"2020","journal-title":"Eur J Endocrinol"},{"issue":"6","key":"10.1016\/j.jot.2024.05.004_bib6","doi-asserted-by":"crossref","first-page":"731","DOI":"10.1016\/j.jbspin.2019.01.012","article-title":"X-linked hypophosphatemia: management and treatment prospects","volume":"86","author":"Lambert","year":"2019","journal-title":"Joint Bone Spine"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib7","doi-asserted-by":"crossref","first-page":"94","DOI":"10.1007\/s002230010045","article-title":"A controlled study of the effects of alendronate in a growing mouse model of osteogenesis imperfecta","volume":"69","author":"Camacho","year":"2001","journal-title":"Calcif Tissue Int"},{"issue":"9419","key":"10.1016\/j.jot.2024.05.004_bib8","doi-asserted-by":"crossref","first-page":"1427","DOI":"10.1016\/S0140-6736(04)16101-1","article-title":"Skeletal effects and functional outcome with olpadronate in children with osteogenesis imperfecta: a 2-year randomised placebo-controlled study","volume":"363","author":"Sakkers","year":"2004","journal-title":"Lancet"},{"issue":"5","key":"10.1016\/j.jot.2024.05.004_bib9","doi-asserted-by":"crossref","first-page":"448","DOI":"10.1007\/s00223-013-9770-2","article-title":"Teriparatide treatment in adult patients with osteogenesis imperfecta type I","volume":"93","author":"Gatti","year":"2013","journal-title":"Calcif Tissue Int"},{"issue":"11","key":"10.1016\/j.jot.2024.05.004_bib10","doi-asserted-by":"crossref","first-page":"898","DOI":"10.1016\/S2213-8587(17)30188-2","article-title":"Osteoporosis treatment: recent developments and ongoing challenges","volume":"5","author":"Khosla","year":"2017","journal-title":"Lancet Diabetes Endocrinol"},{"issue":"4","key":"10.1016\/j.jot.2024.05.004_bib11","doi-asserted-by":"crossref","first-page":"1846","DOI":"10.1210\/jc.2009-1671","article-title":"Treatment of X-linked hypophosphatemia with calcitriol and phosphate increases circulating fibroblast growth factor 23 concentrations","volume":"95","author":"Imel","year":"2010","journal-title":"J Clin Endocrinol Metab"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib12","doi-asserted-by":"crossref","first-page":"274","DOI":"10.1210\/er.2017-00220","article-title":"X-linked hypophosphatemia and FGF23-related hypophosphatemic diseases: prospect for new treatment","volume":"39","author":"Kinoshita","year":"2018","journal-title":"Endocr Rev"},{"key":"10.1016\/j.jot.2024.05.004_bib13","article-title":"Management of X-linked hypophosphatemia in adults","volume":"103s","author":"Lecoq","year":"2020","journal-title":"Metabolism"},{"key":"10.1016\/j.jot.2024.05.004_bib14","article-title":"Clinical and genetic characteristics of 153 Chinese patients with X-linked hypophosphatemia","volume":"9","author":"Lin","year":"2021","journal-title":"Front Cell Dev Biol"},{"issue":"8","key":"10.1016\/j.jot.2024.05.004_bib15","doi-asserted-by":"crossref","first-page":"e3241","DOI":"10.1210\/clinem\/dgac296","article-title":"Effect of Burosumab compared with conventional therapy on younger vs older children with X-linked hypophosphatemia","volume":"107","author":"Ward","year":"2022","journal-title":"J Clin Endocrinol Metab"},{"issue":"10189","key":"10.1016\/j.jot.2024.05.004_bib16","doi-asserted-by":"crossref","first-page":"2416","DOI":"10.1016\/S0140-6736(19)30654-3","article-title":"Burosumab versus conventional therapy in children with X-linked hypophosphataemia: a randomised, active-controlled, open-label, phase 3 trial","volume":"393","author":"Imel","year":"2019","journal-title":"Lancet"},{"issue":"1","key":"10.1016\/j.jot.2024.05.004_bib17","doi-asserted-by":"crossref","first-page":"R13","DOI":"10.1530\/EC-13-0103","article-title":"Therapeutic management of hypophosphatemic rickets from infancy to adulthood","volume":"3","author":"Linglart","year":"2014","journal-title":"Endocr Connect"},{"issue":"7","key":"10.1016\/j.jot.2024.05.004_bib18","doi-asserted-by":"crossref","first-page":"435","DOI":"10.1038\/s41581-019-0152-5","article-title":"Clinical practice recommendations for the diagnosis and management of X-linked hypophosphataemia","volume":"15","author":"Haffner","year":"2019","journal-title":"Nat Rev Nephrol"},{"issue":"1","key":"10.1016\/j.jot.2024.05.004_bib19","doi-asserted-by":"crossref","DOI":"10.3390\/genes14010080","article-title":"The variant p.Ala84Pro is causative of X-linked hypophosphatemic rickets: possible relationship with Burosumab swinging response in adults","volume":"14","author":"Zagari","year":"2022","journal-title":"Genes (Basel)"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib20","doi-asserted-by":"crossref","first-page":"813","DOI":"10.1210\/clinem\/dgab729","article-title":"Sustained efficacy and safety of Burosumab, a monoclonal antibody to FGF23, in children with X-linked hypophosphatemia","volume":"107","author":"Linglart","year":"2022","journal-title":"J Clin Endocrinol Metab"},{"key":"10.1016\/j.jot.2024.05.004_bib21","doi-asserted-by":"crossref","DOI":"10.1155\/2022\/7902046","article-title":"Serum sclerostin and its association with bone turnover marker in metabolic bone diseases","volume":"2022","author":"Chen","year":"2022","journal-title":"Dis Markers"},{"issue":"5","key":"10.1016\/j.jot.2024.05.004_bib22","doi-asserted-by":"crossref","first-page":"E920","DOI":"10.1210\/jc.2013-3852","article-title":"Circulating sclerostin in children and young adults with heritable bone disorders","volume":"99","author":"Palomo","year":"2014","journal-title":"J Clin Endocrinol Metab"},{"key":"10.1016\/j.jot.2024.05.004_bib23","doi-asserted-by":"crossref","first-page":"23","DOI":"10.1016\/j.bone.2014.11.011","article-title":"SPR4-peptide alters bone metabolism of normal and HYP mice","volume":"72","author":"Zelenchuk","year":"2015","journal-title":"Bone"},{"issue":"6","key":"10.1016\/j.jot.2024.05.004_bib24","doi-asserted-by":"crossref","first-page":"591","DOI":"10.1007\/s00223-019-00526-z","article-title":"Elevated bone remodeling markers of CTX and P1NP in addition to sclerostin in patients with X-linked hypophosphatemia: a cross-sectional controlled study","volume":"104","author":"Hansen","year":"2019","journal-title":"Calcif Tissue Int"},{"issue":"1","key":"10.1016\/j.jot.2024.05.004_bib25","doi-asserted-by":"crossref","first-page":"73","DOI":"10.1002\/jbmr.1717","article-title":"Sclerostin antibody improves skeletal parameters in a Brtl\/+ mouse model of osteogenesis imperfecta","volume":"28","author":"Sinder","year":"2013","journal-title":"J Bone Miner Res"},{"issue":"8","key":"10.1016\/j.jot.2024.05.004_bib26","doi-asserted-by":"crossref","first-page":"2097","DOI":"10.1007\/s00198-014-2737-y","article-title":"Adult Brtl\/+ mouse model of osteogenesis imperfecta demonstrates anabolic response to sclerostin antibody treatment with increased bone mass and strength","volume":"25","author":"Sinder","year":"2014","journal-title":"Osteoporos Int"},{"key":"10.1016\/j.jot.2024.05.004_bib27","doi-asserted-by":"crossref","first-page":"115","DOI":"10.1016\/j.bone.2014.10.012","article-title":"Rapidly growing Brtl\/+ mouse model of osteogenesis imperfecta improves bone mass and strength with sclerostin antibody treatment","volume":"71","author":"Sinder","year":"2015","journal-title":"Bone"},{"key":"10.1016\/j.jot.2024.05.004_bib28","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1016\/j.bone.2019.04.011","article-title":"Sclerostin antibody reduces long bone fractures in the oim\/oim model of osteogenesis imperfecta","volume":"124","author":"Cardinal","year":"2019","journal-title":"Bone"},{"issue":"5","key":"10.1016\/j.jot.2024.05.004_bib29","doi-asserted-by":"crossref","first-page":"494","DOI":"10.1007\/s00223-019-00655-5","article-title":"Sclerostin-antibody treatment decreases fracture rates in axial Skeleton and improves the skeletal phenotype in growing oim\/oim mice","volume":"106","author":"Cardinal","year":"2020","journal-title":"Calcif Tissue Int"},{"key":"10.1016\/j.jot.2024.05.004_bib30","doi-asserted-by":"crossref","DOI":"10.1016\/j.bone.2021.116201","article-title":"Sclerostin antibody improves phosphate metabolism hormones, bone formation rates, and bone mass in adult Hyp mice","volume":"154","author":"Carpenter","year":"2022","journal-title":"Bone"},{"issue":"5","key":"10.1016\/j.jot.2024.05.004_bib31","doi-asserted-by":"crossref","first-page":"1030","DOI":"10.1002\/jbmr.2776","article-title":"Sclerostin antibody treatment improves the bone phenotype of Crtap(-\/-) mice, a model of recessive osteogenesis imperfecta","volume":"31","author":"Grafe","year":"2016","journal-title":"J Bone Miner Res"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib32","doi-asserted-by":"crossref","first-page":"596","DOI":"10.1002\/jbmr.3923","article-title":"Sclerostin antibody treatment increases bone mass and normalizes circulating phosphate levels in growing Hyp mice","volume":"35","author":"Carpenter","year":"2020","journal-title":"J Bone Miner Res"},{"issue":"9","key":"10.1016\/j.jot.2024.05.004_bib33","doi-asserted-by":"crossref","first-page":"3183","DOI":"10.1210\/jc.2017-02163","article-title":"A phase III randomized placebo-controlled trial to evaluate efficacy and safety of romosozumab in men with osteoporosis","volume":"103","author":"Lewiecki","year":"2018","journal-title":"J Clin Endocrinol Metab"},{"issue":"15","key":"10.1016\/j.jot.2024.05.004_bib34","doi-asserted-by":"crossref","first-page":"1417","DOI":"10.1056\/NEJMoa1708322","article-title":"Romosozumab or alendronate for fracture prevention in women with osteoporosis","volume":"377","author":"Saag","year":"2017","journal-title":"N Engl J Med"},{"key":"10.1016\/j.jot.2024.05.004_bib35","doi-asserted-by":"crossref","first-page":"191","DOI":"10.1016\/j.bone.2019.01.022","article-title":"Aortic aneurysm\/dissection and osteogenesis imperfecta: four new families and review of the literature","volume":"121","author":"Balasubramanian","year":"2019","journal-title":"Bone"},{"key":"10.1016\/j.jot.2024.05.004_bib36","doi-asserted-by":"crossref","first-page":"149","DOI":"10.1016\/j.ijcard.2015.06.001","article-title":"Heart disease in patients with osteogenesis imperfecta - a systematic review","volume":"196","author":"Ashournia","year":"2015","journal-title":"Int J Cardiol"},{"issue":"5","key":"10.1016\/j.jot.2024.05.004_bib37","doi-asserted-by":"crossref","first-page":"1439","DOI":"10.1016\/0003-4975(95)00706-Q","article-title":"Osteogenesis imperfecta and cardiovascular diseases","volume":"60","author":"Wong","year":"1995","journal-title":"Ann Thorac Surg"},{"issue":"6","key":"10.1016\/j.jot.2024.05.004_bib38","doi-asserted-by":"crossref","first-page":"1077","DOI":"10.1007\/s00467-018-4180-3","article-title":"Risk of cardiovascular involvement in pediatric patients with X-linked hypophosphatemia","volume":"34","author":"Hern\u00e1ndez-Fr\u00edas","year":"2019","journal-title":"Pediatr Nephrol"},{"issue":"8","key":"10.1016\/j.jot.2024.05.004_bib39","doi-asserted-by":"crossref","first-page":"2450","DOI":"10.1210\/jcem.82.8.4181","article-title":"Cardiovascular abnormalities in patients with X-linked hypophosphatemia","volume":"82","author":"Nehgme","year":"1997","journal-title":"J Clin Endocrinol Metab"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib40","doi-asserted-by":"crossref","first-page":"344","DOI":"10.4274\/jcrpe.galenos.2021.2020.0287","article-title":"Left ventricular hypertrophy in patients with X-linked hypophosphataemia","volume":"14","author":"Castellano-Martinez","year":"2022","journal-title":"J Clin Res Pediatr Endocrinol"},{"issue":"1","key":"10.1016\/j.jot.2024.05.004_bib41","doi-asserted-by":"crossref","first-page":"4241","DOI":"10.1038\/s41467-022-31997-8","article-title":"Targeting loop3 of sclerostin preserves its cardiovascular protective action and promotes bone formation","volume":"13","author":"Yu","year":"2022","journal-title":"Nat Commun"},{"issue":"13","key":"10.1016\/j.jot.2024.05.004_bib42","doi-asserted-by":"crossref","first-page":"5645","DOI":"10.7150\/thno.63177","article-title":"Therapeutic aptamer targeting sclerostin loop3 for promoting bone formation without increasing cardiovascular risk in osteogenesis imperfecta mice","volume":"12","author":"Wang","year":"2022","journal-title":"Theranostics"},{"key":"10.1016\/j.jot.2024.05.004_bib43","article-title":"Osteogenesis imperfecta","volume":"3","author":"Marini","year":"2017","journal-title":"Nat Rev Dis Prim"},{"issue":"10028","key":"10.1016\/j.jot.2024.05.004_bib44","doi-asserted-by":"crossref","first-page":"1657","DOI":"10.1016\/S0140-6736(15)00728-X","article-title":"Osteogenesis imperfecta","volume":"387","author":"Forlino","year":"2016","journal-title":"Lancet"},{"issue":"5\u20136","key":"10.1016\/j.jot.2024.05.004_bib45","first-page":"485","article-title":"Safety and efficacy of a 1-year treatment with zoledronic acid compared with pamidronate in children with osteogenesis imperfecta","volume":"25","author":"Barros","year":"2012","journal-title":"J Pediatr Endocrinol Metab"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib46","doi-asserted-by":"crossref","first-page":"355","DOI":"10.1210\/jc.2010-0636","article-title":"Alendronate for the treatment of pediatric osteogenesis imperfecta: a randomized placebo-controlled study","volume":"96","author":"Ward","year":"2011","journal-title":"J Clin Endocrinol Metab"},{"issue":"5","key":"10.1016\/j.jot.2024.05.004_bib47","doi-asserted-by":"crossref","first-page":"660","DOI":"10.1203\/00006450-200211000-00010","article-title":"Alendronate treatment for infants with osteogenesis imperfecta: demonstration of efficacy in a mouse model","volume":"52","author":"McCarthy","year":"2002","journal-title":"Pediatr Res"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib48","doi-asserted-by":"crossref","first-page":"137","DOI":"10.1016\/j.ocl.2013.01.001","article-title":"Bisphosphonate therapy and atypical fractures","volume":"44","author":"Saleh","year":"2013","journal-title":"Orthop Clin N Am"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib49","doi-asserted-by":"crossref","first-page":"277","DOI":"10.1111\/j.1601-0825.2007.01381.x","article-title":"Updates on bisphosphonates and potential pathobiology of bisphosphonate-induced jaw osteonecrosis","volume":"14","author":"Sarin","year":"2008","journal-title":"Oral Dis"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib50","doi-asserted-by":"crossref","first-page":"182","DOI":"10.7326\/M22-0684","article-title":"Effectiveness and safety of treatments to prevent fractures in people with low bone mass or primary osteoporosis: a living systematic review and network meta-analysis for the American college of physicians","volume":"176","author":"Ayers","year":"2023","journal-title":"Ann Intern Med"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib51","doi-asserted-by":"crossref","first-page":"534","DOI":"10.1111\/apa.14154","article-title":"Systematic review of the effect of denosumab on children with osteogenesis imperfecta showed inconsistent findings","volume":"107","author":"Li","year":"2018","journal-title":"Acta Paediatr"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib52","doi-asserted-by":"crossref","first-page":"491","DOI":"10.1172\/JCI71101","article-title":"Evaluation of teriparatide treatment in adults with osteogenesis imperfecta","volume":"124","author":"Orwoll","year":"2014","journal-title":"J Clin Invest"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib53","doi-asserted-by":"crossref","first-page":"312","DOI":"10.1080\/01926230252929882","article-title":"Skeletal changes in rats given daily subcutaneous injections of recombinant human parathyroid hormone (1-34) for 2 years and relevance to human safety","volume":"30","author":"Vahle","year":"2002","journal-title":"Toxicol Pathol"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib54","doi-asserted-by":"crossref","first-page":"237","DOI":"10.1359\/jbmr.2003.18.2.237","article-title":"Positive linear growth and bone responses to growth hormone treatment in children with types III and IV osteogenesis imperfecta: high predictive value of the carboxyterminal propeptide of type I procollagen","volume":"18","author":"Marini","year":"2003","journal-title":"J Bone Miner Res"},{"issue":"10","key":"10.1016\/j.jot.2024.05.004_bib55","article-title":"Recombinant growth hormone therapy for X-linked hypophosphatemia in children","volume":"10","author":"Smith","year":"2021","journal-title":"Cochrane Database Syst Rev"},{"issue":"10","key":"10.1016\/j.jot.2024.05.004_bib56","doi-asserted-by":"crossref","first-page":"2297","DOI":"10.1002\/jbmr.2198","article-title":"Targeting the LRP5 pathway improves bone properties in a mouse model of osteogenesis imperfecta","volume":"29","author":"Jacobsen","year":"2014","journal-title":"J Bone Miner Res"},{"issue":"1","key":"10.1016\/j.jot.2024.05.004_bib57","doi-asserted-by":"crossref","first-page":"48","DOI":"10.1038\/s41413-021-00170-0","article-title":"The WNT1(G177C) mutation specifically affects skeletal integrity in a mouse model of osteogenesis imperfecta type XV","volume":"9","author":"Vollersen","year":"2021","journal-title":"Bone Res"},{"issue":"32","key":"10.1016\/j.jot.2024.05.004_bib58","doi-asserted-by":"crossref","first-page":"26464","DOI":"10.1074\/jbc.M112.350108","article-title":"Characterization of the interaction of sclerostin with the low density lipoprotein receptor-related protein (LRP) family of Wnt co-receptors","volume":"287","author":"Holdsworth","year":"2012","journal-title":"J Biol Chem"},{"issue":"8","key":"10.1016\/j.jot.2024.05.004_bib59","doi-asserted-by":"crossref","DOI":"10.1098\/rsob.160120","article-title":"The sclerostin-neutralizing antibody AbD09097 recognizes an epitope adjacent to sclerostin's binding site for the Wnt co-receptor LRP6","volume":"6","author":"Boschert","year":"2016","journal-title":"Open Biol"},{"issue":"17","key":"10.1016\/j.jot.2024.05.004_bib60","doi-asserted-by":"crossref","first-page":"6899","DOI":"10.1074\/jbc.RA118.006769","article-title":"Lrp4 expression by adipocytes and osteoblasts differentially impacts sclerostin's endocrine effects on body composition and glucose metabolism","volume":"294","author":"Kim","year":"2019","journal-title":"J Biol Chem"},{"issue":"1","key":"10.1016\/j.jot.2024.05.004_bib61","doi-asserted-by":"crossref","first-page":"160","DOI":"10.1016\/j.bbrc.2009.01.062","article-title":"NMR structure of the Wnt modulator protein Sclerostin","volume":"380","author":"Weidauer","year":"2009","journal-title":"Biochem Biophys Res Commun"},{"issue":"10","key":"10.1016\/j.jot.2024.05.004_bib62","doi-asserted-by":"crossref","first-page":"1433","DOI":"10.1016\/j.str.2011.07.005","article-title":"Wnt antagonists bind through a short peptide to the first \u03b2-propeller domain of LRP5\/6","volume":"19","author":"Bourhis","year":"2011","journal-title":"Structure"},{"key":"10.1016\/j.jot.2024.05.004_bib63","article-title":"Sclerostin loop3-LRP4 interaction required by sclerostin for antagonizing Wnt signaling and osteogenic potential in osteoblasts","volume":"FRI-468","author":"Wang","year":"2023","journal-title":"ASBMR 2023 Annual Meeting"},{"issue":"11","key":"10.1016\/j.jot.2024.05.004_bib64","doi-asserted-by":"crossref","first-page":"3487","DOI":"10.1073\/pnas.1419714112","article-title":"Lrp4 in osteoblasts suppresses bone formation and promotes osteoclastogenesis and bone resorption","volume":"112","author":"Xiong","year":"2015","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"48","key":"10.1016\/j.jot.2024.05.004_bib65","doi-asserted-by":"crossref","first-page":"E5187","DOI":"10.1073\/pnas.1413828111","article-title":"Disruption of Lrp4 function by genetic deletion or pharmacological blockade increases bone mass and serum sclerostin levels","volume":"111","author":"Chang","year":"2014","journal-title":"Proc Natl Acad Sci U S A"},{"key":"10.1016\/j.jot.2024.05.004_bib66","doi-asserted-by":"crossref","first-page":"205","DOI":"10.1016\/j.isci.2019.09.023","article-title":"Lrp4 mediates bone homeostasis and mechanotransduction through interaction with sclerostin in vivo","volume":"20","author":"Bullock","year":"2019","journal-title":"iScience"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib67","doi-asserted-by":"crossref","first-page":"113","DOI":"10.1007\/s00018-022-04127-2","article-title":"Competitive blocking of LRP4-sclerostin binding interface strongly promotes bone anabolic functions","volume":"79","author":"Katchkovsky","year":"2022","journal-title":"Cell Mol Life Sci"},{"key":"10.1016\/j.jot.2024.05.004_bib68","doi-asserted-by":"crossref","first-page":"120","DOI":"10.1016\/j.bone.2016.04.011","article-title":"Bone mineral properties in growing Col1a2(+\/G610C) mice, an animal model of osteogenesis imperfecta","volume":"87","author":"Masci","year":"2016","journal-title":"Bone"},{"key":"10.1016\/j.jot.2024.05.004_bib69","doi-asserted-by":"crossref","first-page":"222","DOI":"10.1016\/j.bone.2016.01.001","article-title":"Effect of anti-sclerostin therapy and osteogenesis imperfecta on tissue-level properties in growing and adult mice while controlling for tissue age","volume":"84","author":"Sinder","year":"2016","journal-title":"Bone"},{"issue":"4","key":"10.1016\/j.jot.2024.05.004_bib70","doi-asserted-by":"crossref","first-page":"681","DOI":"10.1016\/j.bone.2007.12.215","article-title":"Role of genetic background in determining phenotypic severity throughout postnatal development and at peak bone mass in Col1a2 deficient mice (oim)","volume":"42","author":"Carleton","year":"2008","journal-title":"Bone"},{"issue":"10","key":"10.1016\/j.jot.2024.05.004_bib71","doi-asserted-by":"crossref","DOI":"10.3390\/biom11101493","article-title":"Osteogenesis imperfecta: current and prospective therapies","volume":"11","author":"Botor","year":"2021","journal-title":"Biomolecules"},{"issue":"6","key":"10.1016\/j.jot.2024.05.004_bib72","doi-asserted-by":"crossref","first-page":"1412","DOI":"10.1002\/jbmr.2177","article-title":"First mouse model for combined osteogenesis imperfecta and Ehlers-Danlos syndrome","volume":"29","author":"Chen","year":"2014","journal-title":"J Bone Miner Res"},{"key":"10.1016\/j.jot.2024.05.004_bib73","doi-asserted-by":"crossref","first-page":"182","DOI":"10.1016\/j.bone.2014.06.015","article-title":"Effect of sclerostin antibody treatment in a mouse model of severe osteogenesis imperfecta","volume":"66","author":"Roschger","year":"2014","journal-title":"Bone"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib74","doi-asserted-by":"crossref","first-page":"291","DOI":"10.1016\/j.cell.2006.08.039","article-title":"CRTAP is required for prolyl 3- hydroxylation and mutations cause recessive osteogenesis imperfecta","volume":"127","author":"Morello","year":"2006","journal-title":"Cell"},{"issue":"15","key":"10.1016\/j.jot.2024.05.004_bib75","doi-asserted-by":"crossref","first-page":"4035","DOI":"10.1093\/hmg\/ddu117","article-title":"The swaying mouse as a model of osteogenesis imperfecta caused by WNT1 mutations","volume":"23","author":"Joeng","year":"2014","journal-title":"Hum Mol Genet"},{"issue":"7","key":"10.1016\/j.jot.2024.05.004_bib76","doi-asserted-by":"crossref","first-page":"2678","DOI":"10.1172\/JCI92617","article-title":"Osteocyte-specific WNT1 regulates osteoblast function during bone homeostasis","volume":"127","author":"Joeng","year":"2017","journal-title":"J Clin Invest"},{"issue":"4","key":"10.1016\/j.jot.2024.05.004_bib77","doi-asserted-by":"crossref","first-page":"539","DOI":"10.1093\/hmg\/6.4.539","article-title":"Distribution of mutations in the PEX gene in families with X-linked hypophosphataemic rickets (HYP)","volume":"6","author":"Rowe","year":"1997","journal-title":"Hum Mol Genet"},{"issue":"6","key":"10.1016\/j.jot.2024.05.004_bib78","doi-asserted-by":"crossref","DOI":"10.1002\/jbm4.10744","article-title":"Asia-pacific consensus recommendations on X-linked hypophosphatemia: diagnosis, multidisciplinary management, and transition from pediatric to adult care","volume":"7","author":"Munns","year":"2023","journal-title":"JBMR Plus"},{"issue":"18","key":"10.1016\/j.jot.2024.05.004_bib79","doi-asserted-by":"crossref","first-page":"1023","DOI":"10.1056\/NEJM198010303031802","article-title":"Bone response to phosphate salts, ergocalciferol, and calcitriol in hypophosphatemic vitamin D-resistant rickets","volume":"303","author":"Glorieux","year":"1980","journal-title":"N Engl J Med"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib80","doi-asserted-by":"crossref","first-page":"223","DOI":"10.1007\/s00467-010-1705-9","article-title":"Age-related stature and linear body segments in children with X-linked hypophosphatemic rickets","volume":"26","author":"Zivi\u010dnjak","year":"2011","journal-title":"Pediatr Nephrol"},{"issue":"12","key":"10.1016\/j.jot.2024.05.004_bib81","doi-asserted-by":"crossref","first-page":"E2097","DOI":"10.1210\/jc.2011-0399","article-title":"Three-year growth hormone treatment in short children with X-linked hypophosphatemic rickets: effects on linear growth and body disproportion","volume":"96","author":"\u017divi\u010dnjak","year":"2011","journal-title":"J Clin Endocrinol Metab"},{"issue":"15","key":"10.1016\/j.jot.2024.05.004_bib82","doi-asserted-by":"crossref","first-page":"6199","DOI":"10.1073\/pnas.1221255110","article-title":"Sclerostin alters serum vitamin D metabolite and fibroblast growth factor 23 concentrations and the urinary excretion of calcium","volume":"110","author":"Ryan","year":"2013","journal-title":"Proc Natl Acad Sci U S A"},{"issue":"11","key":"10.1016\/j.jot.2024.05.004_bib83","doi-asserted-by":"crossref","first-page":"1879","DOI":"10.1359\/jbmr.090509","article-title":"Therapeutic effects of anti-FGF23 antibodies in hypophosphatemic rickets\/osteomalacia","volume":"24","author":"Aono","year":"2009","journal-title":"J Bone Miner Res"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib84","doi-asserted-by":"crossref","first-page":"596","DOI":"10.1002\/jbmr.3923","article-title":"Sclerostin antibody treatment increases bone mass and normalizes circulating phosphate levels in growing Hyp mice","volume":"35","author":"Carpenter","year":"2020","journal-title":"J Bone Miner Res"},{"issue":"1","key":"10.1016\/j.jot.2024.05.004_bib85","doi-asserted-by":"crossref","first-page":"66","DOI":"10.1007\/s00223-021-00823-6","article-title":"Sclerostin directly stimulates osteocyte synthesis of fibroblast growth factor-23","volume":"109","author":"Ito","year":"2021","journal-title":"Calcif Tissue Int"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib86","doi-asserted-by":"crossref","first-page":"R23","DOI":"10.1530\/JME-20-0178","article-title":"FGF23 signalling and physiology","volume":"66","author":"Ho","year":"2021","journal-title":"J Mol Endocrinol"},{"issue":"1","key":"10.1016\/j.jot.2024.05.004_bib87","doi-asserted-by":"crossref","first-page":"407","DOI":"10.1073\/pnas.0902006107","article-title":"Isolated C-terminal tail of FGF23 alleviates hypophosphatemia by inhibiting FGF23-FGFR-Klotho complex formation","volume":"107","author":"Goetz","year":"2010","journal-title":"Proc Natl Acad Sci U S A"},{"key":"10.1016\/j.jot.2024.05.004_bib88","unstructured":"Atlas THP. Gene expression of sclerostin. n.d. Available at: https:\/\/www.proteinatlas.org\/ENSG00000167941-SOST\/tissue. Accessed 2022."},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib89","doi-asserted-by":"crossref","first-page":"495","DOI":"10.1038\/sj.ki.5000148","article-title":"The renal Na+\/phosphate cotransporter NaPi-IIa is internalized via the receptor-mediated endocytic route in response to parathyroid hormone","volume":"69","author":"Bacic","year":"2006","journal-title":"Kidney Int"},{"issue":"12","key":"10.1016\/j.jot.2024.05.004_bib90","doi-asserted-by":"crossref","first-page":"9821","DOI":"10.1007\/s11033-020-05780-1","article-title":"Vitamin D receptor (VDR) and metabolizing enzymes CYP27B1 and CYP24A1 in breast cancer","volume":"47","author":"Voutsadakis","year":"2020","journal-title":"Mol Biol Rep"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib91","doi-asserted-by":"crossref","first-page":"213","DOI":"10.1007\/s00198-022-06523-7","article-title":"Role of Wnt signaling and sclerostin in bone and as therapeutic targets in skeletal disorders","volume":"34","author":"Marini","year":"2023","journal-title":"Osteoporos Int"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib92","doi-asserted-by":"crossref","first-page":"192","DOI":"10.1034\/j.1399-0004.2003.00036.x","article-title":"The natural history of sclerosteosis","volume":"63","author":"Hamersma","year":"2003","journal-title":"Clin Genet"},{"issue":"12","key":"10.1016\/j.jot.2024.05.004_bib93","doi-asserted-by":"crossref","first-page":"2804","DOI":"10.1002\/jbmr.474","article-title":"Patients with sclerosteosis and disease carriers: human models of the effect of sclerostin on bone turnover","volume":"26","author":"van Lierop","year":"2011","journal-title":"J Bone Miner Res"},{"issue":"3","key":"10.1016\/j.jot.2024.05.004_bib94","doi-asserted-by":"crossref","first-page":"577","DOI":"10.1086\/318811","article-title":"Bone dysplasia sclerosteosis results from loss of the SOST gene product, a novel cystine knot-containing protein","volume":"68","author":"Brunkow","year":"2001","journal-title":"Am J Hum Genet"},{"key":"10.1016\/j.jot.2024.05.004_bib95","article-title":"Clinical-functional features of individuals with Osteogenesis Imperfecta and Ehlers-Danlos syndromes: a scoping review of assessment tools and ICF model","volume":"64","author":"Labanca","year":"2023","journal-title":"Musculoskelet Sci Pract"},{"issue":"12","key":"10.1016\/j.jot.2024.05.004_bib96","doi-asserted-by":"crossref","first-page":"2043","DOI":"10.1007\/s11255-016-1379-8","article-title":"Sclerostin as a new key factor in vascular calcification in chronic kidney disease stages 3 and 4","volume":"48","author":"Lv","year":"2016","journal-title":"Int Urol Nephrol"},{"issue":"24","key":"10.1016\/j.jot.2024.05.004_bib97","first-page":"8822","article-title":"Serum sclerostin as a potential novel biomarker for heart valve calcification in patients with chronic kidney disease","volume":"22","author":"Ji","year":"2018","journal-title":"Eur Rev Med Pharmacol Sci"},{"issue":"6","key":"10.1016\/j.jot.2024.05.004_bib98","doi-asserted-by":"crossref","first-page":"737","DOI":"10.1093\/ckj\/sfv077","article-title":"Independent association between serum sclerostin levels and carotid artery atherosclerosis in prevalent haemodialysis patients","volume":"8","author":"Kirkpantur","year":"2015","journal-title":"Clin Kidney J"},{"issue":"6","key":"10.1016\/j.jot.2024.05.004_bib99","doi-asserted-by":"crossref","first-page":"1667","DOI":"10.2337\/dc12-1691","article-title":"Atherosclerotic disease in type 2 diabetes is associated with an increase in sclerostin levels","volume":"36","author":"Morales-Santana","year":"2013","journal-title":"Diabetes Care"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib100","doi-asserted-by":"crossref","first-page":"206","DOI":"10.1016\/j.yexcr.2016.06.007","article-title":"WNT\/\u03b2-catenin signaling promotes VSMCs to osteogenic transdifferentiation and calcification through directly modulating Runx2 gene expression","volume":"345","author":"Cai","year":"2016","journal-title":"Exp Cell Res"},{"issue":"4","key":"10.1016\/j.jot.2024.05.004_bib101","doi-asserted-by":"crossref","first-page":"687","DOI":"10.1002\/jbmr.4503","article-title":"Sclerostin protects against vascular calcification development in mice","volume":"37","author":"De Mar\u00e9","year":"2022","journal-title":"J Bone Miner Res"},{"key":"10.1016\/j.jot.2024.05.004_bib102","doi-asserted-by":"crossref","DOI":"10.1016\/j.metabol.2023.155427","article-title":"Macrophagic sclerostin loop2-ApoER2 interaction required by sclerostin for suppressing inflammatory responses","volume":"142","author":"Wang","year":"2023","journal-title":"Metab, Clin Exp"},{"key":"10.1016\/j.jot.2024.05.004_bib103","series-title":"Proc the world congress insulin resistance diabetes & cardiovascular diseases (WCIRDC)","article-title":"Macrophagic sclerostin loop2-ApoER2 interaction required by sclerostin for suppressing inflammatory responses","author":"Wang","year":"2022"},{"issue":"7","key":"10.1016\/j.jot.2024.05.004_bib104","doi-asserted-by":"crossref","first-page":"421","DOI":"10.1016\/j.matbio.2004.09.007","article-title":"Homozygous ablation of fibroblast growth factor-23 results in hyperphosphatemia and impaired skeletogenesis, and reverses hypophosphatemia in Phex-deficient mice","volume":"23","author":"Sitara","year":"2004","journal-title":"Matrix Biol"},{"key":"10.1016\/j.jot.2024.05.004_bib105","doi-asserted-by":"crossref","DOI":"10.1016\/j.bone.2022.116602","article-title":"Bone characteristics of autosomal dominant hypophosphatemic rickets patients","volume":"167","author":"Liu","year":"2023","journal-title":"Bone"},{"issue":"2","key":"10.1016\/j.jot.2024.05.004_bib106","doi-asserted-by":"crossref","first-page":"830","DOI":"10.1016\/j.bbrc.2011.12.048","article-title":"The sclerostin-bone protein interactome","volume":"417","author":"Devarajan-Ketha","year":"2012","journal-title":"Biochem Biophys Res Commun"}],"container-title":["Journal of Orthopaedic Translation"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2214031X24000469?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S2214031X24000469?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,5,15]],"date-time":"2026-05-15T15:29:28Z","timestamp":1778858968000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S2214031X24000469"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,7]]},"references-count":106,"alternative-id":["S2214031X24000469"],"URL":"https:\/\/doi.org\/10.1016\/j.jot.2024.05.004","relation":{},"ISSN":["2214-031X"],"issn-type":[{"value":"2214-031X","type":"print"}],"subject":[],"published":{"date-parts":[[2024,7]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Sclerostin inhibition in rare bone diseases: Molecular understanding and therapeutic perspectives","name":"articletitle","label":"Article Title"},{"value":"Journal of Orthopaedic Translation","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.jot.2024.05.004","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2024 The Authors. Published by Elsevier B.V. on behalf of Chinese Speaking Orthopaedic Society.","name":"copyright","label":"Copyright"}]}}