Role of RANKL (TNFSF11)-Dependent Osteopetrosis in the Dental Phenotype of Msx2 Null Mutant Mice
Open accessAbstract
The MSX2 homeoprotein is implicated in all aspects of craniofacial skeletal development. During postnatal growth, MSX2 is expressed in all cells involved in mineralized tissue formation and plays a role in their differentiation and function. Msx2 null (Msx2 (-/-)) mice display complex craniofacial skeleton abnormalities with bone and tooth defects. A moderate form osteopetrotic phenotype is observed, along with decreased expression of RANKL (TNFSF11), the main osteoclast-differentiating factor. In order to elucidate the role of such an osteopetrosis in the Msx2 (-/-) mouse dental phenotype, a bone resorption rescue was performed by mating Msx2 (-/-) mice with a transgenic mouse line overexpressing Rank (Tnfrsf11a). Msx2 (-/-) Rank(Tg) mice had significant improvement in the molar phenotype, while incisor epithelium defects were exacerbated in the enamel area, with formation of massive osteolytic tumors. Although compensation for RANKL loss of function could have potential as a therapy for osteopetrosis, but in Msx2 (-/-) mice, this approach via RANK overexpression in monocyte-derived lineages, amplified latent epithelial tumor development in the peculiar continuously growing incisor.
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Msx2 deficient adult mice show a slight osteopetrosis phenotype in the skull, tibia, and mandible, which could be reversed by RANK overexpression.
“It has also been reported that Msx2 deficient adult mice show a slight osteopetrosis phenotype in the skull, tibia, and mandible, which could be reversed by RANK overexpression24.”
From Targeting Msx2 as a brake in the fusion fate of osteoclasts and an anabolic therapy in pre-clinical models of osteoporosis (2025) · cites this paper as [24]
The Results and Discussion report soft osteopetrosis in the tibia that shifted toward osteopenia with RANK overexpression; they also report partial closure of a skull defect. But the Figure 5 caption says the Msx2-deficient mandible had no major alterations compared with wild type, while RANK overexpression caused marked mandibular bone disruption. The paper therefore does not support reversal of a slight osteopetrosis phenotype across all three sites. Page numbers were not available in our copy. (LLM, conf=0.88)
Experimental — accuracy not yet measuredMsx2 null mutant mice show osteopetrosis in a RANK-dependent manner, suggesting Msx2 might participate in osteoclastogenesis.
“Although Msx2 is generally considered an important regulator of osteogenesis, Msx2 null mutant mice show an osteopetrosis phenotype in a RANK-dependent manner in skeleton24, suggesting that Msx2 might also participate in osteoclastogenesis, and its regulatory effect in the bone environment could be complex.”
From Targeting Msx2 as a brake in the fusion fate of osteoclasts and an anabolic therapy in pre-clinical models of osteoporosis (2025) · cites this paper as [24]
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References 27 · Cited by 15
- Authors
- Castaneda, B., Simon, Y., Ferbus, D., Robert, B., Chesneau, J., Mueller, C., Berdal, A., & Lézot, F.
- Affiliations
- Inserm (FR)
- Centre National de la Recherche Scientifique (FR)
- Corresponding author
- Lézot, Frédéric
- Venue
- PLoS ONE
- Publisher
- Public Library of Science (PLoS)
- Published
- 2013-11-21
- Volume
- 8
- Issue
- 11
- Pages
- e80054
- ISSN
- 1932-6203 (ISSN-L)
- PMC
- PMC3836916
- OpenAlex
- W2079555209
- Cited by
- 15
- References
- 27
- Citations by year
- 2025: 1 · 2024: 1 · 2020: 2 · 2019: 3 · 2018: 3 · 2017: 1 · 2016: 1 · 2015: 2 · 2014: 1
- FWCI
- 0.60
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Licence
- Locations
- PLoS ONE (publishedVersion, cc-by)
- Europe PMC (PubMed Central) (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- HAL (submittedVersion, cc-by)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
- Figshare (submittedVersion, cc-by)
- Greater South Information System
- Funders
- Institut National de la Santé et de la Recherche Médicale
- Institut National Du Cancer
- National Cancer Institute
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Bone Metabolism and Diseases
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › dental development and anomalies
- Health Sciences › Medicine › Rheumatology › Bone and Dental Protein Studies
- Keywords
- Msx2, osteopetrosis, RANKL, dental phenotype, osteoclastogenesis, bone resorption, enamel defects, osteolytic tumor, monocyte-derived cells
- MeSH terms
- Animals, Base Sequence, Osteopetrosis, Phenotype, Tooth, DNA Primers, Mice, Knockout, Homeodomain Proteins, Reverse Transcriptase Polymerase Chain Reaction, Mice, RANK Ligand, X-Ray Microtomography, Real-Time Polymerase Chain Reaction
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