ArticleThe Journal of clinical endocrinology and metabolism2019
Denosumab in Patients With Fibrous Dysplasia Previously Treated With Bisphosphonates.
Article in The Journal of clinical endocrinology and metabolism, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 43 papers, 3 of them syntheses that pooled it.
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Who cites it
43 citing papers in PubMed, 3 syntheses or guidelines pooled it, 72 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
- No overshoot of bone turnover after withdrawal of denosumab treatment of adults with Langerhans cell histiocytosis: a prospective clinical trial.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2025Trial
- RANKL inhibition reduces lesional cellularity and GαBone research · 2024Trial
- Individualized antiresorptive therapy in fibrous dysplasia and McCune-Albright syndrome: A retrospective cohort study.Bone reports · 2026Article
- Intensive, interdisciplinary pain treatment in fibrous dysplasia/McCune-Albright syndrome.JBMR plus · 2026Article
- In vitro characterization of osteoblasts from craniofacial fibrous dysplasia of bone and their impact on bone homeostasis.Orphanet journal of rare diseases · 2026Article
- Denosumab in pediatric patients with fibrous dysplasia/McCune-Albright syndrome: a single-center, open-labeled study.Frontiers in endocrinology · 2026Article
- Multidisciplinary approach and clinical management of McCune-Albright syndrome.Frontiers in endocrinology · 2026Review
- Article
- Bone pain in fibrous dysplasia does not rely on aberrant sensory nerve sprouting or neuroma formation.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2025Article
- Article
- Denosumab usage in atypical cemento-osseous dysplasia involving the entire mandible: a case report.BMC oral health · 2025Article
- Update on the medical management of fibrous dysplasia of the bone.Therapeutic advances in endocrinology and metabolism · 2025Review
- Rebound Hypercalcemia After Denosumab Cessation in Adult Fibrous Dysplasia: A Case Report and Clinical Alert.Case reports in endocrinology · 2025Article
- McCune-Albright Syndrome: A Case From Mauritius.Cureus · 2024Article
- Mazabraud's syndrome in female patients: Two case reports.World journal of orthopedics · 2024Article
- Management of RANKL-mediated Disorders With Denosumab in Children and Adolescents: A Global Expert Guidance Document.The Journal of clinical endocrinology and metabolism · 2024Article
Corrections and comments
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Authors and funding
6 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
contextFibrous dysplasia/McCune-Albright syndrome (FD/MAS) is a rare bone disorder commonly treated with bisphosphonates, but clinical and biochemical responses may be incomplete.
objectiveTo evaluate the efficacy and tolerability of the receptor activator of nuclear factor-κB ligand inhibitor denosumab in the treatment of patients with FD/MAS refractory to bisphosphonate therapy.
designCase series.
settingAcademic center of expertise for rare bone diseases. PATIENTS: Data were collected from 12 consecutive patients with FD/MAS with persistent pain and increased biochemical markers of bone turnover (BTMs) after long-term treatment with bisphosphonates (median, 8.8 years) and were treated with subcutaneous denosumab 60 mg at 3- or 6-month intervals with a follow-up for at least 12 months. MAIN OUTCOME(S): Sustained reduction of BTMs and bone pain.
resultsA 60 mg dose of denosumab once every 3 months, but not once every 6 months, induced a sustained reduction of BTMs. After a median treatment period of 15.5 months (range, 12 to 19) serum alkaline phosphatase activity and propeptide of type 1 procollagen levels were respectively reduced from 212 ± 39.4 IU/L to 79 ± 6.0 IU/L (P = 0.004) and from 346.2 ± 111.1 ng/mL to 55.7 ± 16.6 ng/mL (P = 0.023) and normalized in 70% and 75% of patients, respectively. Although not quantitavely measured, 10 patients reported a reduction in bone pain of whom 6 reported complete elimination of pain. Treatment with denosumab was well tolerated.
conclusionOur results indicate that 60 mg of denosumab every 3 months is a promising, well-tolerated treatment of most patients with FD/MAS refractory to bisphosphonate therapy. These results together with those of previously published case reports provide the necessary background for the design of a larger, controlled study.
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