ArticleJournal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research2015
Neural Crest-Specific TSC1 Deletion in Mice Leads to Sclerotic Craniofacial Bone Lesion.
Article in Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 30 papers.
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Who cites it
30 citing papers in PubMed, 37 citations in OpenAlex.
- Region- and Compartment-Specific Elevation of Bone Mass in Mice Following Tsc1 Deletion in 8-kb Dmp1-Cre-Expressing Cells.Calcified tissue international · 2026Article
- COVID-19 and Congenital Cleft Lip and Palate: A Systematic Review Focusing on Nonsyndromic Neonatal Outcomes.Cureus · 2026Review
- Loss of Tsc1 in Osterix-expressing cells leads to greater bone mass and strength in mice.Bone · 2026Article
- The level of protein in the maternal murine diet modulates the facial appearance of the offspring via mTORC1 signaling.Nature communications · 2024Article
- Off-target activity of the 8 kb Dmp1-Cre results in the deletion of Tsc1 gene in mouse intestinal mesenchyme.Transgenic research · 2023Article
- Article
- Expression of Cre recombinase in chondrocytes causes abnormal craniofacial and skeletal development.Transgenic research · 2022Article
- Skeletal diseases caused by mutations inFrontiers in cell and developmental biology · 2022Article
- Article
- Review
- Article
- DEPTOR in Skeletal Development and Diseases.Frontiers in genetics · 2021Review
- FAK Promotes Early Osteoprogenitor Cell Proliferation by Enhancing mTORC1 Signaling.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2020Article
- Bone tissue and mineral metabolism in hereditary endocrine tumors: clinical manifestations and genetic bases.Orphanet journal of rare diseases · 2020Review
- Autophagy Regulates Craniofacial Bone Acquisition.Calcified tissue international · 2019Article
- Activation of mTORC1 in subchondral bone preosteoblasts promotes osteoarthritis by stimulating bone sclerosis and secretion of CXCL12.Bone research · 2019Article
- Targeting the Wnt/β-catenin pathway in human osteosarcoma cells.Oncotarget · 2018Article
- Mid-facial developmental defects caused by the widely used LacZ reporter gene when expressed in neural crest-derived cells.Transgenic research · 2018Article
- Tsc1 Regulates the Balance Between Osteoblast and Adipocyte Differentiation Through Autophagy/Notch1/β-Catenin Cascade.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2018Article
- mTOR acts as a pivotal signaling hub for neural crest cells during craniofacial development.PLoS genetics · 2018Article
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Authors and funding
7 authors at 4 institutions in 1 country.
Funding
Abstract
Tuberous sclerosis complex (TSC) is an autosomal dominant disorder caused by mutations in either TSC1 or TSC2. TSC has high frequency of osseous manifestations such as sclerotic lesions in the craniofacial region. However, an animal model that replicates TSC craniofacial bone lesions has not yet been described. The roles of Tsc1 and the sequelae of Tsc1 dysfunction in bone are unknown. In this study, we generated a mouse model of TSC with a deletion of Tsc1 in neural crest-derived (NCD) cells that recapitulated the sclerotic craniofacial bone lesions in TSC. Analysis of this mouse model demonstrated that TSC1 deletion led to enhanced mTORC1 signaling in NCD bones and the increase in bone formation is responsible for the aberrantly increased bone mass. Lineage mapping revealed that TSC1 deficient NCD cells overpopulated the NCD bones. Mechanistically, hyperproliferation of osteoprogenitors at an early postnatal stage accounts for the increased osteoblast pool. Intriguingly, early postnatal treatment with rapamycin, an mTORC1 inhibitor, can completely rescue the aberrant bone mass, but late treatment cannot. Our data suggest that enhanced mTOR signaling in NCD cells can increase bone mass through enlargement of the osteoprogenitor pool, which likely explains the sclerotic bone lesion observed in TSC patients.
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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.