ArticleThe American journal of pathology1997
Increased proliferation of osteoblastic cells expressing the activating Gs alpha mutation in monostotic and polyostotic fibrous dysplasia.
Article in The American journal of pathology, 1997. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 50 papers.
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Who cites it
50 citing papers in PubMed, 175 citations in OpenAlex.
- Individualized antiresorptive therapy in fibrous dysplasia and McCune-Albright syndrome: A retrospective cohort study.Bone reports · 2026Article
- In vitro characterization of osteoblasts from craniofacial fibrous dysplasia of bone and their impact on bone homeostasis.Orphanet journal of rare diseases · 2026Article
- The GNAS Gene: Fibrous Dysplasia, McCune-Albright Syndrome, and Skeletal Structure and Function.Genes · 2025Review
- Article
- Diagnostic journey for individuals with fibrous dysplasia / McCune albright syndrome (FD/MAS).Orphanet journal of rare diseases · 2024Article
- A Closer Look at Fibrous Dysplasia Femur Fracture Five-year Follow-up: A Unique Case.Journal of orthopaedic case reports · 2024Article
- A Rare Skeletal Disorder, Fibrous Dysplasia: A Review of Its Pathogenesis and Therapeutic Prospects.International journal of molecular sciences · 2023Review
- Woven bone formation and mineralization by rat mesenchymal stromal cells imply increased expression of the intermediate filament desmin.Frontiers in endocrinology · 2023Article
- Comprehensive management of malocclusion in maxillary fibrous dysplasia: A case report.World journal of clinical cases · 2021Article
- From Stem Cells to Bone-Forming Cells.International journal of molecular sciences · 2021Review
- PET Imaging of Phosphodiesterase-4 Identifies Affected Dysplastic Bone in McCune-Albright Syndrome, a Genetic Mosaic Disorder.Journal of nuclear medicine : official publication, Society of Nuclear Medicine · 2020Article
- Jawbone fibrous dysplasia: retrospective evaluation in a cases series surgically treated and short review of the literature.Acta bio-medica : Atenei Parmensis · 2020Review
- An Analysis of Clinical and Histopathologic Features of Fibrous Dysplasia of the Jaws: A Series of 40 Cases and Review of Literature.Head and neck pathology · 2020Review
- HDAC8, A Potential Therapeutic Target, Regulates Proliferation and Differentiation of Bone Marrow Stromal Cells in Fibrous Dysplasia.Stem cells translational medicine · 2019Article
- Management of Fibrous Dysplasia of Proximal Femur by Internal Fixation Without Grafting: A Retrospective Study of 19 Patients.Journal of the American Academy of Orthopaedic Surgeons. Global research & reviews · 2018Article
- InducedProceedings of the National Academy of Sciences of the United States of America · 2018Article
- Frequency of GNAS R201H substitution mutation in polyostotic fibrous dysplasia: Pyrosequencing analysis in tissue samples with or without decalcification.Scientific reports · 2017Article
- Heterotrimeric G proteins in the control of parathyroid hormone actions.Journal of molecular endocrinology · 2017Review
- Functional and radiological outcomes of a minimally invasive surgical approach to monostotic fibrous dysplasia.World journal of surgical oncology · 2017Article
- Article
Corrections and comments
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
We studied the osteoblastic abnormalities resulting from activating mutation of the Gs alpha gene in two patients with McCune-Albright syndrome and one patient with monostotic fibrous dysplasia. Histomorphometric analysis of dysplastic lesions showed a low number of differentiated osteoblasts along the bone surface and numerous immature alkaline phosphatase-positive mesenchymal cells actively depositing a woven bone matrix. Osteoblastic cells isolated from dysplastic bone lesions expressed a missense mutation in the Gs alpha gene in position 201 and showed increased intracellular basal cyclic adenosine 3',5'-monophosphate levels compared with normal cells isolated from a noninvolved area in the same patient. Cell proliferation evaluated by DNA synthesis was two-fold to threefold greater in osteoblastic cells expressing the mutation compared with normal cells from the same patient and was greater in cells isolated from more severe than less severe fibrotic lesions. In contrast, the synthesis of osteocalcin, a marker of mature osteoblasts, was lower in osteoblastic cells expressing the Gs alpha mutation compared with normal cells from the same patient and was lower in cells isolated from severe compared with less severe fibrotic lesions, indicating that the increased growth in mutated osteoblastic cells was associated with reduced cell differentiation. The results show that activating mutation of Gs alpha in osteoblastic cells leads to constitutive activation of adenylate cyclase, increased cell proliferation, and inappropriate cell differentiation, resulting in overproduction of a disorganized fibrotic bone matrix in polyostotic and monostotic fibrous dysplasia.
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