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

Osteogenesis 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 Am...

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Autores principales:	Rachel Kohler, Carli A Tastad, Amy Creecy, Joseph M Wallace
Formato:	article
Lenguaje:	EN
Publicado:	Public Library of Science (PLoS) 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/705e950b3cc74900a0ef6792bb9987ac
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spelling	oai:doaj.org-article:705e950b3cc74900a0ef6792bb9987ac2021-12-02T20:17:31ZMorphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta.1932-620310.1371/journal.pone.0255315https://doaj.org/article/705e950b3cc74900a0ef6792bb9987ac2021-01-01T00:00:00Zhttps://doi.org/10.1371/journal.pone.0255315https://doaj.org/toc/1932-6203Osteogenesis 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.Rachel KohlerCarli A TastadAmy CreecyJoseph M WallacePublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 16, Iss 8, p e0255315 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Rachel Kohler Carli A Tastad Amy Creecy Joseph M Wallace Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta.
description	Osteogenesis 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.
format	article
author	Rachel Kohler Carli A Tastad Amy Creecy Joseph M Wallace
author_facet	Rachel Kohler Carli A Tastad Amy Creecy Joseph M Wallace
author_sort	Rachel Kohler
title	Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta.
title_short	Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta.
title_full	Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta.
title_fullStr	Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta.
title_full_unstemmed	Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta.
title_sort	morphological and mechanical characterization of bone phenotypes in the amish g610c murine model of osteogenesis imperfecta.
publisher	Public Library of Science (PLoS)
publishDate	2021
url	https://doaj.org/article/705e950b3cc74900a0ef6792bb9987ac
work_keys_str_mv	AT rachelkohler morphologicalandmechanicalcharacterizationofbonephenotypesintheamishg610cmurinemodelofosteogenesisimperfecta AT carliatastad morphologicalandmechanicalcharacterizationofbonephenotypesintheamishg610cmurinemodelofosteogenesisimperfecta AT amycreecy morphologicalandmechanicalcharacterizationofbonephenotypesintheamishg610cmurinemodelofosteogenesisimperfecta AT josephmwallace morphologicalandmechanicalcharacterizationofbonephenotypesintheamishg610cmurinemodelofosteogenesisimperfecta
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Morphological and mechanical characterization of bone phenotypes in the Amish G610C murine model of osteogenesis imperfecta.

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