Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta

dc.contributor.authorKohler, Rachel
dc.contributor.authorTastad, Carli A.
dc.contributor.authorCreecy, Amy
dc.contributor.authorWallace, Joseph M.
dc.contributor.departmentBiomedical Engineering, School of Engineering and Technologyen_US
dc.date.accessioned2023-02-27T12:37:02Z
dc.date.available2023-02-27T12:37:02Z
dc.date.issued2021-08-27
dc.description.abstractOsteogenesis imperfecta (OI) is a hereditary bone disease where gene mutations affect Type I collagen formation resulting in osteopenia and increased fracture risk. There are several established mouse models of OI, but some are severe and result in spontaneous fractures or early animal death. The Amish Col1a2G610C/+ (G610C) mouse model is a newer, moderate OI model that is currently being used in a variety of intervention studies, with differing background strains, sexes, ages, and bone endpoints. This study is a comprehensive mechanical and architectural characterization of bone in G610C mice bred on a C57BL/6 inbred strain and will provide a baseline for future treatment studies. Male and female wild-type (WT) and G610C mice were euthanized at 10 and 16 weeks (n = 13-16). Harvested tibiae, femora, and L4 vertebrae were scanned via micro-computed tomography and analyzed for cortical and trabecular architectural properties. Femora and tibiae were then mechanically tested to failure. G610C mice had less bone but more highly mineralized cortical and trabecular tissue than their sex- and age-matched WT counterparts, with cortical cross-sectional area, thickness, and mineral density, and trabecular bone volume, mineral density, spacing, and number all differing significantly as a function of genotype (2 Way ANOVA with main effects of sex and genotype at each age). In addition, mechanical yield force, ultimate force, displacement, strain, and toughness were all significantly lower in G610C vs. WT, highlighting a brittle phenotype. This characterization demonstrates that despite being a moderate OI model, the Amish G610C mouse model maintains a distinctly brittle phenotype and is well-suited for use in future intervention studies.en_US
dc.eprint.versionFinal published versionen_US
dc.identifier.citationKohler R, Tastad CA, Creecy A, Wallace JM. Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta. PLoS One. 2021;16(8):e0255315. Published 2021 Aug 27. doi:10.1371/journal.pone.0255315en_US
dc.identifier.urihttps://hdl.handle.net/1805/31490
dc.language.isoen_USen_US
dc.publisherPLOSen_US
dc.relation.isversionof10.1371/journal.pone.0255315en_US
dc.relation.journalPLOS ONEen_US
dc.rightsAttribution 4.0 International*
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/*
dc.sourcePMCen_US
dc.subjectOsteogenesis imperfectaen_US
dc.subjectX-Ray microtomographyen_US
dc.subjectBone diseaseen_US
dc.titleMorphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfectaen_US
dc.typeArticleen_US
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