ArticleJournal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research2014
How tough is brittle bone? Investigating osteogenesis imperfecta in mouse bone.
Article in Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 60 papers.
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Who cites it
60 citing papers in PubMed, 143 citations in OpenAlex.
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- Molecular dynamics reveals how calcium drives hetero- versus homotrimerization of type I collagen.Biophysical journal · 2026Article
- Molecular drivers of osteogenesis imperfecta: a cellular and extracellular collagen disease.Clinical science (London, England : 1979) · 2025Review
- Improvement of bone properties in children with osteogenesis imperfecta after pamidronate: a bone biopsy study.JBMR plus · 2025Article
- Characterization of a Novel Col1a1Calcified tissue international · 2025Article
- Bone Quality and Mineralization and Effects of Treatment in Osteogenesis Imperfecta.Calcified tissue international · 2024Review
- Bone health: Quality versus quantity.Journal of the Pediatric Orthopaedic Society of North America · 2024Article
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- Bone Material Properties in Bone Diseases Affecting Children.Current osteoporosis reports · 2023Review
- Col1A-2 Mutation in Osteogenesis Imperfecta Mice Contributes to Long Bone Fragility by Modifying Cell-Matrix Organization.International journal of molecular sciences · 2023Article
- In Vivo Assessment of Skin Surface Pattern: Exploring Its Potential as an Indicator of Bone Biomechanical Properties.Bioengineering (Basel, Switzerland) · 2023Article
- Review
- Collagen piezoelectricity in osteogenesis imperfecta and its role in intrafibrillar mineralization.Communications biology · 2022Article
- An Update on Animal Models of Osteogenesis Imperfecta.Calcified tissue international · 2022Review
- Collagen (I) homotrimer potentiates the osteogenesis imperfecta (oim) mutant allele and reduces survival in male mice.Disease models & mechanisms · 2022Article
- Biomechanical, Microstructural and Material Properties of Tendon and Bone in the Young Oim Mice Model of Osteogenesis Imperfecta.International journal of molecular sciences · 2022Article
- Article
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Authors and funding
10 authors at 4 institutions in 2 countries.
Funding
Abstract
The multiscale hierarchical structure of bone is naturally optimized to resist fractures. In osteogenesis imperfecta, or brittle bone disease, genetic mutations affect the quality and/or quantity of collagen, dramatically increasing bone fracture risk. Here we reveal how the collagen defect results in bone fragility in a mouse model of osteogenesis imperfecta (oim), which has homotrimeric α1(I) collagen. At the molecular level, we attribute the loss in toughness to a decrease in the stabilizing enzymatic cross-links and an increase in nonenzymatic cross-links, which may break prematurely, inhibiting plasticity. At the tissue level, high vascular canal density reduces the stable crack growth, and extensive woven bone limits the crack-deflection toughening during crack growth. This demonstrates how modifications at the bone molecular level have ramifications at larger length scales affecting the overall mechanical integrity of the bone; thus, treatment strategies have to address multiscale properties in order to regain bone toughness. In this regard, findings from the heterozygous oim bone, where defective as well as normal collagen are present, suggest that increasing the quantity of healthy collagen in these bones helps to recover toughness at the multiple length scales.
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