ArticleProceedings of the National Academy of Sciences of the United States of America2010
A mouse model of osteochondromagenesis from clonal inactivation of Ext1 in chondrocytes.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2010. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 63 papers.
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Who cites it
63 citing papers in PubMed, 126 citations in OpenAlex.
- Article
- Genotype and phenotype correlation analysis in 27 families with multiple osteochondroma and validation by ATDC5 chondrocyte models.Bone & joint research · 2025Article
- Free glutaraldehyde gelatin microsphere loaded mesenchymal stem cells alleviate osteoarthritis by promotingTheranostics · 2025Article
- Analysis of the Actions of RARγ Agonists on Growing Osteochondromas in a Mouse Model.International journal of molecular sciences · 2024Article
- Article
- Osteochondroma formation is independent of heparanase expression as revealed in a mouse model of hereditary multiple exostoses.Journal of orthopaedic research : official publication of the Orthopaedic Research Society · 2022Article
- Role of nuclear factor of activated T cells in chondrogenesis osteogenesis and osteochondroma formation.Journal of endocrinological investigation · 2022Article
- Genetic and functional analyses detect an EXT1 splicing pathogenic variant in a Chinese hereditary multiple exostosis (HME) family.Molecular genetics & genomic medicine · 2022Article
- Physiology and Pathophysiology of Heparan Sulfate in Animal Models: Its Biosynthesis and Degradation.International journal of molecular sciences · 2022Review
- Osteochondroma Pathogenesis: Mouse Models and Mechanistic Insights into Interactions with Retinoid Signaling.The American journal of pathology · 2021Review
- Heparan Sulfate Deficiency in Cartilage: Enhanced BMP-Sensitivity, Proteoglycan Production and an Anti-Apoptotic Expression Signature after Loading.International journal of molecular sciences · 2021Article
- Mutation spectrum of EXT1 and EXT2 in the Saudi patients with hereditary multiple exostoses.Orphanet journal of rare diseases · 2021Article
- Site-Specific Recombination with Inverted Target Sites: A Cautionary Tale of Dicentric and Acentric Chromosomes.Genetics · 2020Article
- An altered heparan sulfate structure in the articular cartilage protects against osteoarthritis.Osteoarthritis and cartilage · 2020Article
- Article
- Non-ossifying fibroma: A RAS-MAPK driven benign bone neoplasm.The Journal of pathology · 2019Article
- A novel splice mutation induces exon skipping of the EXT1 gene in patients with hereditary multiple exostoses.International journal of oncology · 2019Article
- Hereditary Multiple Exostoses: Current Insights.Orthopedic research and reviews · 2019Review
- c-Fos induces chondrogenic tumor formation in immortalized human mesenchymal progenitor cells.Scientific reports · 2018Article
- The pathogenic roles of heparan sulfate deficiency in hereditary multiple exostoses.Matrix biology : journal of the International Society for Matrix Biology · 2018Review
3 more citing papers are in PubMed but not listed here.
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12 authors at 6 institutions in 4 countries.
Funding
Abstract
We report a mouse model of multiple osteochondromas (MO), an autosomal dominant disease in humans, also known as multiple hereditary exostoses (MHE or HME) and characterized by the formation of cartilage-capped osseous growths projecting from the metaphyses of endochondral bones. The pathogenesis of these osteochondromas has remained unclear. Mice heterozygous for Ext1 or Ext2, modeling the human genotypes that cause MO, occasionally develop solitary osteochondroma-like structures on ribs [Lin et al. (2000) Dev Biol 224(2):299-311; Stickens et al. (2005) Development 132(22):5055-5068]. Rather than model the germ-line genotype, we modeled the chimeric tissue genotype of somatic loss of heterozygosity (LOH), by conditionally inactivating Ext1 via head-to-head loxP sites and temporally controlled Cre-recombinase in chondrocytes. These mice faithfully recapitulate the human phenotype of multiple metaphyseal osteochondromas. We also confirm homozygous disruption of Ext1 in osteochondroma chondrocytes and their origin in proliferating physeal chondrocytes. These results explain prior modeling failures with the necessity for somatic LOH in a developmentally regulated cell type.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.