{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T06:41:25Z","timestamp":1783579285634,"version":"3.55.0"},"reference-count":52,"publisher":"Oxford University Press (OUP)","issue":"10","license":[{"start":{"date-parts":[[2014,2,12]],"date-time":"2014-02-12T00:00:00Z","timestamp":1392163200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"name":"Osteogenesis Imperfecta Foundation Michael Geisman Fellowship"},{"name":"Pediatric Endocrinology Society Research Fellowship"},{"name":"NIAMS\/NIH","award":["AR37694"],"award-info":[{"award-number":["AR37694"]}]},{"name":"NIAMS\/NIH","award":["AR37318"],"award-info":[{"award-number":["AR37318"]}]},{"name":"NIAMS\/NIH","award":["R01AR053237"],"award-info":[{"award-number":["R01AR053237"]}]},{"name":"NIAMS\/NIH","award":["R21AR062326"],"award-info":[{"award-number":["R21AR062326"]}]},{"name":"NICHD\/NIH","award":["HD070394"],"award-info":[{"award-number":["HD070394"]}]},{"DOI":"10.13039\/100000011","name":"Howard Hughes Medical Institute","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000011","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Melissa Cummings and Animal Resources at Children's Hospital"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2014,10,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>\u2003<\/jats:title>\n                    <jats:p>The cell surface receptor low-density lipoprotein receptor-related protein 5 (LRP5) is a key regulator of bone mass and bone strength. Heterozygous missense mutations in LRP5 cause autosomal dominant high bone mass (HBM) in humans by reducing binding to LRP5 by endogenous inhibitors, such as sclerostin (SOST). Mice heterozygous for a knockin allele (Lrp5p.A214V) that is orthologous to a human HBM-causing mutation have increased bone mass and strength. Osteogenesis imperfecta (OI) is a skeletal fragility disorder predominantly caused by mutations that affect type I collagen. We tested whether the LRP5 pathway can be used to improve bone properties in animal models of OI. First, we mated Lrp5+\/p.A214V mice to Col1a2+\/p.G610C mice, which model human type IV OI. We found that Col1a2+\/p.G610C;Lrp5+\/p.A214V offspring had significantly increased bone mass and strength compared to Col1a2+\/p.G610C;Lrp5+\/+ littermates. The improved bone properties were not a result of altered mRNA expression of type I collagen or its chaperones, nor were they due to changes in mutant type I collagen secretion. Second, we treated Col1a2+\/p.G610C mice with a monoclonal antibody that inhibits sclerostin activity (Scl-Ab). We found that antibody-treated mice had significantly increased bone mass and strength compared to vehicle-treated littermates. These findings indicate increasing bone formation, even without altering bone collagen composition, may benefit patients with OI. \u00a9 2014 American Society for Bone and Mineral Research.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/jbmr.2198","type":"journal-article","created":{"date-parts":[[2014,2,12]],"date-time":"2014-02-12T07:33:13Z","timestamp":1392190393000},"page":"2297-2306","source":"Crossref","is-referenced-by-count":77,"title":["Targeting the LRP5 Pathway Improves Bone Properties in a Mouse Model of Osteogenesis Imperfecta"],"prefix":"10.1093","volume":"29","author":[{"given":"Christina M","family":"Jacobsen","sequence":"first","affiliation":[{"name":"Department of PediatricsHarvard Medical SchoolBostonMAUSA"},{"name":"Division of GeneticsBoston Children's HospitalBostonMAUSA"},{"name":"Division of EndocrinologyBoston Children's HospitalBostonMAUSA"},{"name":"Orthopaedic Research LaboratoriesDepartment of Orthopaedic Surgery, Boston Children's HospitalBostonMAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lauren A","family":"Barber","sequence":"additional","affiliation":[{"name":"Orthopaedic Research LaboratoriesDepartment of Orthopaedic Surgery, Boston Children's HospitalBostonMAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ugur M","family":"Ayturk","sequence":"additional","affiliation":[{"name":"Department of GeneticsHarvard Medical SchoolBostonMAUSA"},{"name":"Orthopaedic Research LaboratoriesDepartment of Orthopaedic Surgery, Boston Children's HospitalBostonMAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heather J","family":"Roberts","sequence":"additional","affiliation":[{"name":"Orthopaedic Research LaboratoriesDepartment of Orthopaedic Surgery, Boston Children's HospitalBostonMAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lauren E","family":"Deal","sequence":"additional","affiliation":[{"name":"Department of Anatomy and Cell BiologyIndiana UniversityIndianapolisINUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marissa A","family":"Schwartz","sequence":"additional","affiliation":[{"name":"Orthopaedic Research LaboratoriesDepartment of Orthopaedic Surgery, Boston Children's HospitalBostonMAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"MaryAnn","family":"Weis","sequence":"additional","affiliation":[{"name":"Department of Orthopedics and Sports MedicineUniversity of WashingtonSeattleWAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Eyre","sequence":"additional","affiliation":[{"name":"Department of Orthopedics and Sports MedicineUniversity of WashingtonSeattleWAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Zurakowski","sequence":"additional","affiliation":[{"name":"Department of AnesthesiaChildren's Hospital BostonBostonMAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexander G","family":"Robling","sequence":"additional","affiliation":[{"name":"Department of Anatomy and Cell BiologyIndiana UniversityIndianapolisINUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew L","family":"Warman","sequence":"additional","affiliation":[{"name":"Howard Hughes Medical InstituteBostonMAUSA"},{"name":"Department of GeneticsHarvard Medical SchoolBostonMAUSA"},{"name":"Orthopaedic Research LaboratoriesDepartment of Orthopaedic Surgery, Boston Children's HospitalBostonMAUSA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2014,2,12]]},"reference":[{"issue":"6","key":"2024020318483633300_jbmr2198-bib-0001","doi-asserted-by":"crossref","first-page":"439","DOI":"10.1007\/s00223-012-9588-3","article-title":"Recent advances in osteogenesis imperfecta","volume":"90","author":"Cundy","year":"2012","journal-title":"Calcif Tissue Int."},{"key":"2024020318483633300_jbmr2198-bib-0002","doi-asserted-by":"crossref","first-page":"475","DOI":"10.1146\/annurev-genet-110711-155608","article-title":"Recessively inherited forms of osteogenesis imperfecta","volume":"46","author":"Byers","year":"2012","journal-title":"Annu Rev Genet."},{"issue":"14","key":"2024020318483633300_jbmr2198-bib-0003","doi-asserted-by":"crossref","first-page":"947","DOI":"10.1056\/NEJM199810013391402","article-title":"Cyclic administration of pamidronate in children with severe osteogenesis imperfecta","volume":"339","author":"Glorieux","year":"1998","journal-title":"N Engl J Med."},{"issue":"3","key":"2024020318483633300_jbmr2198-bib-0004","doi-asserted-by":"crossref","first-page":"573","DOI":"10.1542\/peds.111.3.573","article-title":"Intravenous bisphosphonate therapy in children with osteogenesis imperfecta","volume":"111","author":"Falk","year":"2003","journal-title":"Pediatrics."},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0005","first-page":"38","article-title":"Pamidronate treatment of children with moderate-to-severe osteogenesis imperfecta: a note of caution","volume":"71","author":"Alharbi","year":"2009","journal-title":"Horm Res."},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0006","doi-asserted-by":"crossref","first-page":"285","DOI":"10.1007\/s00198-011-1658-2","article-title":"Risedronate in adults with osteogenesis imperfecta type I: increased bone mineral density and decreased bone turnover, but high fracture rate persists","volume":"23","author":"Bradbury","year":"2012","journal-title":"Osteoporos Int."},{"issue":"4","key":"2024020318483633300_jbmr2198-bib-0007","first-page":"CD005088","article-title":"Bisphosphonate therapy for osteogenesis imperfecta","author":"Phillipi","year":"2008","journal-title":"Cochrane Database Syst Rev."},{"issue":"11","key":"2024020318483633300_jbmr2198-bib-0008","doi-asserted-by":"crossref","first-page":"2267","DOI":"10.1002\/jbmr.253","article-title":"Atypical subtrochanteric and diaphyseal femoral fractures: report of a task force of the American Society for Bone and Mineral Research","volume":"25","author":"Shane","year":"2010","journal-title":"J Bone Miner Res."},{"issue":"13","key":"2024020318483633300_jbmr2198-bib-0009","doi-asserted-by":"crossref","first-page":"1480","DOI":"10.1001\/jama.2010.1360","article-title":"Atypical fractures as a potential complication of long-term bisphosphonate therapy","volume":"304","author":"Sellmeyer","year":"2010","journal-title":"JAMA."},{"issue":"4","key":"2024020318483633300_jbmr2198-bib-0010","doi-asserted-by":"crossref","first-page":"513","DOI":"10.1016\/S0092-8674(01)00571-2","article-title":"LDL receptor-related protein 5 (LRP5) affects bone accrual and eye development","volume":"107","author":"Gong","year":"2001","journal-title":"Cell."},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0011","doi-asserted-by":"crossref","first-page":"11","DOI":"10.1086\/338450","article-title":"A mutation in the LDL receptor-related protein 5 gene results in the autosomal dominant high-bone-mass trait","volume":"70","author":"Little","year":"2002","journal-title":"Am J Hum Genet."},{"issue":"20","key":"2024020318483633300_jbmr2198-bib-0012","doi-asserted-by":"crossref","first-page":"1513","DOI":"10.1056\/NEJMoa013444","article-title":"High bone density due to a mutation in LDL-receptor-related protein 5","volume":"346","author":"Boyden","year":"2002","journal-title":"N Engl J Med."},{"issue":"50","key":"2024020318483633300_jbmr2198-bib-0013","doi-asserted-by":"crossref","first-page":"38276","DOI":"10.1074\/jbc.M609509200","article-title":"LRP5 mutations linked to high bone mass diseases cause reduced LRP5 binding and inhibition by SOST","volume":"281","author":"Semenov","year":"2006","journal-title":"J Biol Chem."},{"issue":"6","key":"2024020318483633300_jbmr2198-bib-0014","doi-asserted-by":"crossref","first-page":"445","DOI":"10.1007\/s00223-008-9130-9","article-title":"The binding between sclerostin and LRP5 is altered by DKK1 and by high-bone mass LRP5 mutations","volume":"82","author":"Balemans","year":"2008","journal-title":"Calcif Tissue Int."},{"issue":"11","key":"2024020318483633300_jbmr2198-bib-0015","doi-asserted-by":"crossref","first-page":"1738","DOI":"10.1359\/jbmr.060810","article-title":"Bone density ligand, Sclerostin, directly interacts with LRP5 but not LRP5G171V to modulate Wnt activity","volume":"21","author":"Ellies","year":"2006","journal-title":"J Bone Miner Res."},{"issue":"6","key":"2024020318483633300_jbmr2198-bib-0016","doi-asserted-by":"crossref","first-page":"684","DOI":"10.1038\/nm.2388","article-title":"Lrp5 functions in bone to regulate bone mass","volume":"17","author":"Cui","year":"2011","journal-title":"Nat Med."},{"issue":"3","key":"2024020318483633300_jbmr2198-bib-0017","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."},{"issue":"5","key":"2024020318483633300_jbmr2198-bib-0018","doi-asserted-by":"crossref","first-page":"537","DOI":"10.1093\/hmg\/10.5.537","article-title":"Increased bone density in sclerosteosis is due to the deficiency of a novel secreted protein (SOST)","volume":"10","author":"Balemans","year":"2001","journal-title":"Hum Mol Genet."},{"issue":"2","key":"2024020318483633300_jbmr2198-bib-0019","doi-asserted-by":"crossref","first-page":"144","DOI":"10.1002\/ajmg.10401","article-title":"A 52-kb deletion in the SOST-MEOX1 intergenic region on 17q12-q21 is associated with van Buchem disease in the Dutch population","volume":"110","author":"Staehling-Hampton","year":"2002","journal-title":"Am J Med Genet."},{"issue":"2","key":"2024020318483633300_jbmr2198-bib-0020","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1136\/jmg.39.2.91","article-title":"Identification of a 52 kb deletion downstream of the SOST gene in patients with van Buchem disease","volume":"39","author":"Balemans","year":"2002","journal-title":"J Med Genet."},{"issue":"11","key":"2024020318483633300_jbmr2198-bib-0021","doi-asserted-by":"crossref","first-page":"2412","DOI":"10.1002\/jbmr.135","article-title":"Sclerostin antibody treatment enhances metaphyseal bone healing in rats","volume":"25","author":"Agholme","year":"2010","journal-title":"J Bone Miner Res."},{"issue":"5","key":"2024020318483633300_jbmr2198-bib-0022","doi-asserted-by":"crossref","first-page":"1012","DOI":"10.1002\/jbmr.307","article-title":"Inhibition of sclerostin by monoclonal antibody enhances bone healing and improves bone density and strength of nonfractured bones","volume":"26","author":"Ominsky","year":"2011","journal-title":"J Bone Miner Res."},{"issue":"5","key":"2024020318483633300_jbmr2198-bib-0023","doi-asserted-by":"crossref","first-page":"948","DOI":"10.1002\/jbmr.14","article-title":"Two doses of sclerostin antibody in cynomolgus monkeys increases bone formation, bone mineral density, and bone strength","volume":"25","author":"Ominsky","year":"2010","journal-title":"J Bone Miner Res."},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0024","doi-asserted-by":"crossref","first-page":"19","DOI":"10.1002\/jbmr.173","article-title":"Single-dose, placebo-controlled, randomized study of AMG 785, a sclerostin monoclonal antibody","volume":"26","author":"Padhi","year":"2011","journal-title":"J Bone Miner Res."},{"issue":"12","key":"2024020318483633300_jbmr2198-bib-0025","doi-asserted-by":"crossref","first-page":"2647","DOI":"10.1002\/jbmr.182","article-title":"Inhibition of sclerostin by monoclonal antibody increases bone formation, bone mass, and bone strength in aged male rats","volume":"25","author":"Li","year":"2010","journal-title":"J Bone Miner Res."},{"issue":"4","key":"2024020318483633300_jbmr2198-bib-0026","doi-asserted-by":"crossref","first-page":"578","DOI":"10.1359\/jbmr.081206","article-title":"Sclerostin antibody treatment increases bone formation, bone mass, and bone strength in a rat model of postmenopausal osteoporosis","volume":"24","author":"Li","year":"2009","journal-title":"J Bone Miner Res."},{"issue":"2","key":"2024020318483633300_jbmr2198-bib-0027","doi-asserted-by":"crossref","first-page":"247","DOI":"10.1359\/jbmr.090720","article-title":"Variable bone fragility associated with an Amish COL1A2 variant and a knock-in mouse model","volume":"25","author":"Daley","year":"2010","journal-title":"J Bone Miner Res."},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0028","doi-asserted-by":"crossref","first-page":"52","DOI":"10.2144\/00291bm09","article-title":"Preparation of PCR-quality mouse genomic DNA with hot sodium hydroxide and tris (HotSHOT)","volume":"29","author":"Truett","year":"2000","journal-title":"Biotechniques."},{"issue":"33","key":"2024020318483633300_jbmr2198-bib-0029","doi-asserted-by":"crossref","first-page":"23698","DOI":"10.1074\/jbc.M601000200","article-title":"The Wnt co-receptor LRP5 is essential for skeletal mechanotransduction but not for the anabolic bone response to parathyroid hormone treatment","volume":"281","author":"Sawakami","year":"2006","journal-title":"J Biol Chem."},{"key":"2024020318483633300_jbmr2198-bib-0030","doi-asserted-by":"crossref","first-page":"2495","DOI":"10.1099\/00221287-146-10-2495","article-title":"Comparison of proteins expressed by Pseudomonas aeruginosa strains representing initial and chronic isolates from a cystic fibrosis patient: an analysis by 2-D gel electrophoresis and capillary column liquid chromatography-tandem mass spectrometry","volume":"146","author":"Hanna","year":"2000","journal-title":"Microbiology."},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0031","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1016\/j.ymeth.2008.01.002","article-title":"Advances in collagen cross-link analysis","volume":"45","author":"Eyre","year":"2008","journal-title":"Methods."},{"issue":"10","key":"2024020318483633300_jbmr2198-bib-0032","doi-asserted-by":"crossref","first-page":"2081","DOI":"10.1002\/jbmr.1946","article-title":"An RNA-seq protocol to identify mRNA expression changes in mouse diaphyseal bone: applications in mice with bone property altering Lrp5 mutations","volume":"28","author":"Ayturk","year":"2013","journal-title":"J Bone Miner Res."},{"issue":"18","key":"2024020318483633300_jbmr2198-bib-0033","doi-asserted-by":"crossref","first-page":"2518","DOI":"10.1093\/bioinformatics\/btr427","article-title":"Comparative analysis of RNA-Seq alignment algorithms and the RNA-Seq unified mapper (RUM)","volume":"27","author":"Grant","year":"2011","journal-title":"Bioinformatics."},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0034","doi-asserted-by":"crossref","first-page":"139","DOI":"10.1093\/bioinformatics\/btp616","article-title":"edgeR: a Bioconductor package for differential expression analysis of digital gene expression data","volume":"26","author":"Robinson","year":"2010","journal-title":"Bioinformatics."},{"key":"2024020318483633300_jbmr2198-bib-0035","doi-asserted-by":"crossref","first-page":"591","DOI":"10.1093\/biomet\/52.3-4.591","article-title":"An analysis of variance test for normality","volume":"52","author":"Shapiro","year":"1965","journal-title":"Biometrika."},{"key":"2024020318483633300_jbmr2198-bib-0036","volume-title":"Biometry: the principles and practice of statistics in biological research","author":"Sokal","year":"1995","edition":"3rd ed"},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0037","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":"20","key":"2024020318483633300_jbmr2198-bib-0038","doi-asserted-by":"crossref","first-page":"19883","DOI":"10.1074\/jbc.M413274200","article-title":"Sclerostin binds to LRP5\/6 and antagonizes canonical Wnt signaling","volume":"280","author":"Li","year":"2005","journal-title":"J Biol Chem."},{"issue":"29","key":"2024020318483633300_jbmr2198-bib-0039","doi-asserted-by":"crossref","first-page":"26770","DOI":"10.1074\/jbc.M504308200","article-title":"SOST is a ligand for LRP5\/LRP6 and a Wnt signaling inhibitor","volume":"280","author":"Semenov","year":"2005","journal-title":"J Biol Chem."},{"issue":"12","key":"2024020318483633300_jbmr2198-bib-0040","doi-asserted-by":"crossref","first-page":"2033","DOI":"10.1359\/jbmr.040907","article-title":"Decreased BMD and limb deformities in mice carrying mutations in both Lrp5 and Lrp6","volume":"19","author":"Holmen","year":"2004","journal-title":"J Bone Miner Res."},{"issue":"2","key":"2024020318483633300_jbmr2198-bib-0041","doi-asserted-by":"crossref","first-page":"490","DOI":"10.1016\/j.bone.2011.08.005","article-title":"Trabecular bone loss after administration of the second-generation antipsychotic risperidone is independent of weight gain","volume":"50","author":"Motyl","year":"2012","journal-title":"Bone."},{"issue":"1","key":"2024020318483633300_jbmr2198-bib-0042","doi-asserted-by":"crossref","first-page":"50","DOI":"10.1007\/s00223-012-9610-9","article-title":"Early response of bone marrow osteoprogenitors to skeletal unloading and sclerostin antibody","volume":"91","author":"Shahnazari","year":"2012","journal-title":"Calcif Tissue Int."},{"issue":"7","key":"2024020318483633300_jbmr2198-bib-0043","doi-asserted-by":"crossref","first-page":"1531","DOI":"10.1002\/jbmr.1892","article-title":"A mouse model for human osteogenesis imperfecta type VI","volume":"28","author":"Bogan","year":"2013","journal-title":"J Bone Miner Res."},{"issue":"22","key":"2024020318483633300_jbmr2198-bib-0044","doi-asserted-by":"crossref","first-page":"17253","DOI":"10.1074\/jbc.M110.102228","article-title":"Prolyl 3-hydroxylase 1 null mice display abnormalities in fibrillar collagen-rich tissues such as tendons, skin, and bones","volume":"285","author":"Vranka","year":"2010","journal-title":"J Biol Chem."},{"issue":"2","key":"2024020318483633300_jbmr2198-bib-0045","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":"2","key":"2024020318483633300_jbmr2198-bib-0046","doi-asserted-by":"crossref","first-page":"e7","DOI":"10.1371\/journal.pgen.0040007","article-title":"ER stress-mediated apoptosis in a new mouse model of osteogenesis imperfecta","volume":"4","author":"Lisse","year":"2008","journal-title":"PLoS Genet."},{"issue":"8","key":"2024020318483633300_jbmr2198-bib-0047","doi-asserted-by":"crossref","first-page":"803","DOI":"10.1038\/ng1603","article-title":"A deletion in the gene encoding sphingomyelin phosphodiesterase 3 (Smpd3) results in osteogenesis and dentinogenesis imperfecta in the mouse","volume":"37","author":"Aubin","year":"2005","journal-title":"Nat Genet."},{"issue":"53","key":"2024020318483633300_jbmr2198-bib-0048","doi-asserted-by":"crossref","first-page":"37923","DOI":"10.1074\/jbc.274.53.37923","article-title":"Use of the Cre\/lox recombination system to develop a non-lethal knock-in murine model for osteogenesis imperfecta with an alpha1(I) G349C substitution. Variability in phenotype in BrtlIV mice","volume":"274","author":"Forlino","year":"1999","journal-title":"J Biol Chem."},{"issue":"5","key":"2024020318483633300_jbmr2198-bib-0049","doi-asserted-by":"crossref","first-page":"1701","DOI":"10.1073\/pnas.90.5.1701","article-title":"Defective pro alpha 2(I) collagen synthesis in a recessive mutation in mice: a model of human osteogenesis imperfecta","volume":"90","author":"Chipman","year":"1993","journal-title":"Proc Natl Acad Sci U S A."},{"issue":"6060","key":"2024020318483633300_jbmr2198-bib-0050","doi-asserted-by":"crossref","first-page":"365","DOI":"10.1038\/320365a0","article-title":"Retrovirus insertion inactivates mouse alpha 1(I) collagen gene by blocking initiation of transcription","volume":"320","author":"Hartung","year":"1986","journal-title":"Nature."},{"issue":"6","key":"2024020318483633300_jbmr2198-bib-0051","doi-asserted-by":"crossref","first-page":"598","DOI":"10.1002\/humu.21475","article-title":"COL1 C-propeptide cleavage site mutations cause high bone mass osteogenesis imperfecta","volume":"32","author":"Lindahl","year":"2011","journal-title":"Hum Mutat."},{"issue":"5","key":"2024020318483633300_jbmr2198-bib-0052","doi-asserted-by":"crossref","first-page":"849","DOI":"10.1359\/jbmr.081238","article-title":"Alendronate treatment of the brtl osteogenesis imperfecta mouse improves femoral geometry and load response before fracture but decreases predicted material properties and has detrimental effects on osteoblasts and bone formation","volume":"24","author":"Uveges","year":"2009","journal-title":"J Bone Miner Res."}],"container-title":["Journal of Bone and Mineral Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fjbmr.2198","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fjbmr.2198","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/academic.oup.com\/jbmr\/article-pdf\/29\/10\/2297\/56565669\/jbmr2198.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jbmr\/article-pdf\/29\/10\/2297\/56565669\/jbmr2198.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,3,13]],"date-time":"2024-03-13T10:51:39Z","timestamp":1710327099000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jbmr\/article\/29\/10\/2297-2306\/7598934"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2014,2,12]]},"references-count":52,"journal-issue":{"issue":"10","published-online":{"date-parts":[[2014,2,12]]},"published-print":{"date-parts":[[2014,10,1]]}},"URL":"https:\/\/doi.org\/10.1002\/jbmr.2198","relation":{"has-review":[{"id-type":"doi","id":"10.3410\/f.718302207.793491899","asserted-by":"object"}]},"ISSN":["0884-0431","1523-4681"],"issn-type":[{"value":"0884-0431","type":"print"},{"value":"1523-4681","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2014,10,1]]},"published":{"date-parts":[[2014,2,12]]}}}