Mutation of the sequestosome 1 (p62) gene increases osteoclastogenesis but does not induce Paget disease
Open accessAbstract
Paget disease is the most exaggerated example of abnormal bone remodeling, with the primary cellular abnormality in the osteoclast. Mutations in the p62 (sequestosome 1) gene occur in one-third of patients with familial Paget disease and in a minority of patients with sporadic Paget disease, with the P392L amino acid substitution being the most commonly observed mutation. However, it is unknown how p62(P392L) mutation contributes to the development of this disease. To determine the effects of p62(P392L) expression on osteoclasts in vitro and in vivo, we introduced either the p62(P392L) or WT p62 gene into normal osteoclast precursors and targeted p62(P392L) expression to the osteoclast lineage in transgenic mice. p62(P392L)-transduced osteoclast precursors were hyperresponsive to receptor activator of NF-kappaB ligand (RANKL) and TNF-alpha and showed increased NF-kappaB signaling but did not demonstrate increased 1,25-(OH)(2)D(3) responsivity, TAF(II)-17 expression, or nuclear number per osteoclast. Mice expressing p62(P392L) developed increased osteoclast numbers and progressive bone loss, but osteoblast numbers were not coordinately increased, as is seen in Paget disease. These results indicate that p62(P392L) expression on osteoclasts is not sufficient to induce the full pagetic phenotype but suggest that p62 mutations cause a predisposition to the development of Paget disease by increasing the sensitivity of osteoclast precursors to osteoclastogenic cytokines.
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Overexpression of SQSTM1 variants carrying disease-associated mutations in the UBA domain exhibits a PDB-like skeletal disorder and impaired spatial learning and long-term memory.
“On the other hand, overexpression of SQSTM1 variants carrying disease-associated mutations in the UBA domain exhibit a PDB-like skeletal disorder [138,139] as well as impaired spatial learning and long-term memory [140].”
From Autophagy and ALS: mechanistic insights and therapeutic implications (2022) · cites this paper as [139]
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- Authors
- Kurihara, N., Hiruma, Y., Zhou, H., Subler, M. A., Dempster, D. W., Singer, F. R., Reddy, S. V., Gruber, H. E., Windle, J. J., & Roodman, G. D.
- Affiliations
- VA Pittsburgh Healthcare System (US)
- University of Pittsburgh (US)
- Virginia Commonwealth University (US)
- Columbia University (US)
- Medical University of South Carolina (US)
- Carolinas Medical Center (US)
- Corresponding author
- Kurihara, Noriyoshi
- ORCID
- Venue
- Journal of Clinical Investigation
- Published
- 2007-01-02
- Volume
- 117
- Issue
- 1
- Pages
- 133-142
- ISSN
- 0021-9738 (ISSN-L)
- PMC
- PMC1705822
- OpenAlex
- W2116217447
- Cited by
- 127
- References
- 25
- Citations by year
- 2025: 2 · 2024: 1 · 2023: 4 · 2022: 3 · 2021: 5 · 2020: 2 · 2019: 5 · 2018: 5 · 2017: 4 · 2016: 4 · 2015: 5 · 2014: 9 · 2013: 8 · 2012: 7
- FWCI
- 4.48
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- Journal of Clinical Investigation (publishedVersion)
- PubMed Central (publishedVersion)
- PubMed (publishedVersion)
- Funders
- University of Pittsburgh
- National Institutes of Health (UL1TR000005, CA16059)
- VA Pittsburgh Healthcare System
- National Cancer Institute (P30CA16059, P30CA016059)
- National Institute of Arthritis and Musculoskeletal and Skin Diseases (P01AR049363)
- National Center for Advancing Translational Sciences
- Medical Research and Materiel Command
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Bone Metabolism and Diseases
- Health Sciences › Medicine › Oncology › Bone health and treatments
- Health Sciences › Medicine › Rheumatology › Osteomyelitis and Bone Disorders Research
- Keywords
- p62, Paget disease, osteoclastogenesis, osteoclasts, bone remodeling, RANKL, NF-κB signaling, TNF-α, 1,25-dihydroxyvitamin D3, transgenic mice, bone loss, osteoclast precursors
- MeSH terms
- Sequestosome-1 Protein, Calcitriol, Cell Division, Humans, Mutation, Osteitis Deformans, Osteoclasts, Proteins, Tumor Necrosis Factor-alpha, Gene Transfer Techniques, Genetic Predisposition to Disease, Adaptor Proteins, Signal Transducing, RANK Ligand
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