ArticleThe Journal of clinical endocrinology and metabolism2021
Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[18F]F PET-CT in Patients With Fibrous Dysplasia/McCune-Albright Syndrome.
Article in The Journal of clinical endocrinology and metabolism, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 16 papers, 3 of them syntheses that pooled it.
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Who cites it
16 citing papers in PubMed, 3 syntheses or guidelines pooled it, 25 citations in OpenAlex.
- The use of denosumab in rare bone diseases in adults: a systematic review from the ECTS Rare Bone Disease Action Group.The Journal of clinical endocrinology and metabolism · 2026Pooled it
- Comparison of traditional and new treatments for fibrous dysplasia: a systematic review and meta-analysis.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2026Pooled it
- Efficacy of antiresorptive agents in fibrous dysplasia and McCune Albright syndrome, a systematic review and meta-analysis.Reviews in endocrine & metabolic disorders · 2023Pooled it
- Safety and Efficacy of Moderate-dose Denosumab in Fibrous Dysplasia: Observational Results From a Phase 2 Clinical Trial.The Journal of clinical endocrinology and metabolism · 2025Trial
- Individualized antiresorptive therapy in fibrous dysplasia and McCune-Albright syndrome: A retrospective cohort study.Bone reports · 2026Article
- Denosumab in pediatric patients with fibrous dysplasia/McCune-Albright syndrome: a single-center, open-labeled study.Frontiers in endocrinology · 2026Article
- Article
- Rebound Hypercalcemia After Denosumab Cessation in Adult Fibrous Dysplasia: A Case Report and Clinical Alert.Case reports in endocrinology · 2025Article
- [Biomolecules · 2024Review
- Structural and molecular imaging-based characterization of soft tissue and vascular calcification in hyperphosphatemic familial tumoral calcinosis.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2024Article
- Safety and Efficacy of Denosumab for Fibrous Dysplasia of Bone.The New England journal of medicine · 2023Article
- Long Bone Fractures in Fibrous Dysplasia/McCune-Albright Syndrome: Prevalence, Natural History, and Risk Factors.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2022Article
- Successful treatment with denosumab for pelvic fibrous dysplasia: A case report and review of the literature.Medicine · 2021Review
- Safety of therapy with and withdrawal from denosumab in fibrous dysplasia and McCune-Albright syndrome: an observational study.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2021Observational
- Review
- Regression of fibrous dysplasia in response to denosumab therapy: A report of two cases.Bone reports · 2021Article
Corrections and comments
- Erratum issued
Authors and funding
7 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
contextThe correlation between fibrous dysplasia/McCune-Albright syndrome (FD/MAS) skeletal disease burden on Na[18F]F positron emission tomography-computed tomography (PET-CT) and serum bone turnover markers (BTMs) was recently described. The effect of treatment on lesional fluoride burden in FD/MAS is unknown.
objectiveTo investigate treatment response measurements in patients with FD/MAS who underwent Na[18F]F-PET-CT and treatment with antiresorptives.
methodsObservational case series at an academic center of expertise for rare bone diseases. Fifteen consecutive patients were observed with FD/MAS with baseline and follow-up Na[18F]F-PET-CT parameters of healthy bone and FD lesions, BTMs, and pain scores at start of denosumab (n = 8) treatment and non-denosumab patients (n = 7). On Na[18F]F-PET-CT the volumetric measures of FD burden (fluoride tumor volume [FTV]) and "fraction affected skeleton" (FAS) represented the portion of the skeleton affected. This was correlated with BTMs and pain.
resultsDisease activity decreased significantly, with FTV 361 cm3 to 97 cm3 (P = .018) in denosumab-treated patients, but not in non-denosumab patients (P = .249). Serum P1NP and alkaline phosphatase (ALP) decreased significantly: 82 ng/mL vs 55 ng/mL (P = .023) and 119 IU/L vs 84 IU/L (P = .020), respectively. In denosumab-treated patients pain scores improved leading to pain medication reduction. This correlated with lesional uptake, but healthy bone activity did not change. BTMs and FTV correlated positively (P1NP r = 0.730, P < .001; and ALP r = 0.406, P = .006), as did change in BTMs and FTV: P1NP (P = 0.032) and ALP (P = 0.024). FAS strongly correlated with treatment-induced decrease in ALP (P = .027) and P1NP (P = .009).
conclusionNa[18F]F-PET-CT captured treatment-induced lesional changes which correlated with BTMs and pain reduction. Therefore Na[18F]F-PET-CT can be used as an objective local parameter of response to denosumab treatment in FD/MAS.
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