ArticleClinical cases in mineral and bone metabolism : the official journal of the Italian Society of Osteoporosis, Mineral Metabolism, and Skeletal Diseases
Prompt clinical and biochemical response to denosumab in a young adult patient with craniofacial fibrous dysplasia.
Article in Clinical cases in mineral and bone metabolism : the official journal of the Italian Society of Osteoporosis, Mineral Metabolism, and Skeletal Diseases. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers, 1 of them a synthesis that pooled it.
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20 citing papers in PubMed, 1 synthesis or guideline pooled it, 32 citations in OpenAlex.
- 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
- Intensive, interdisciplinary pain treatment in fibrous dysplasia/McCune-Albright syndrome.JBMR plus · 2026Article
- Rebound Hypercalcemia After Denosumab Cessation in Adult Fibrous Dysplasia: A Case Report and Clinical Alert.Case reports in endocrinology · 2025Article
- Phenotyping Pain in Patients With Fibrous Dysplasia/McCune-Albright Syndrome.The Journal of clinical endocrinology and metabolism · 2024Article
- A Rare Skeletal Disorder, Fibrous Dysplasia: A Review of Its Pathogenesis and Therapeutic Prospects.International journal of molecular sciences · 2023Review
- Current concepts in targeted therapies for benign tumors of the jaw - A review of the literature.Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery · 2023Review
- Bone Turnover Markers: Basic Biology to Clinical Applications.Endocrine reviews · 2023Article
- Pharmacological Interventions Targeting Pain in Fibrous Dysplasia/McCune-Albright Syndrome.International journal of molecular sciences · 2023Review
- Fibrous Dysplasia of Temporal Bone.Indian journal of otolaryngology and head and neck surgery : official publication of the Association of Otolaryngologists of India · 2022Article
- Case Report: The Imperfect Association Between Craniofacial Lesion Burden and Pain in Fibrous Dysplasia.Frontiers in neurology · 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
- Denosumab for craniofacial fibrous dysplasia: duration of efficacy and post-treatment effects.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2021Article
- Fibrous Dysplasia/McCune-Albright Syndrome: A Rare, Mosaic Disease of Gα s Activation.Endocrine reviews · 2020Review
- RANKL Inhibition in Fibrous Dysplasia of Bone: A Preclinical Study in a Mouse Model of the Human Disease.Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research · 2019Article
- Use of Denosumab in Children With Osteoclast Bone Dysplasias: Report of Three Cases.JBMR plus · 2019Article
- Clinical and translational pharmacological aspects of the management of fibrous dysplasia of bone.British journal of clinical pharmacology · 2019Review
- Article
- Advances in Models of Fibrous Dysplasia/McCune-Albright Syndrome.Frontiers in endocrinology · 2019Review
- Different clinical presentation and management of temporal bone fibrous dysplasia in children.World journal of surgical oncology · 2018Review
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Authors and funding
9 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundWe report on the clinical and biochemical outcomes in a 20-year-old male suffering from active craniofacial monostotic fibrous dysplasia (MFD) of the left mandible treated with the RANK-L inhibitor, denosumab, following unsatisfactory responses to prior long-term bisphosphonates therapy.
resultsThe patient had been treated over 9 years with pamidronate (cumulative dose of 810 mg) with incomplete control of pain. Following initiation of denosumab 60 mg subcutaneously, bone pain and bone turnover markers (osteocalcin, total and bone alkaline phosphatase and carboxy-terminal cross-linking telopeptide of type I collagen) were monitored over a 27 months period. Few hours after the first administration, the patient demonstrated a complete pain disappearance and after 4 weeks bone turnover markers fell within the normal range. Three months after denosumab initiation the patient reported a pain reactivation that required a second administration, which again led to the pain disappearance. Subsequently, denosumab was administered according to the pain reappearance and the injection was always followed by complete pain relief. However, a gradual shortening of the pain-free interval between administrations was observed, ranging from 90 to 75 days. All bone turnover markers stayed in the lower half of the normal range, even at the moment of pain reappearance, suggesting that the effect of denosumab on pain depends on mechanisms other than bone resorption suppression. No side effects were reported by the patient during the follow-up.
conclusionDenosumab appears to be effective in reducing bone turnover and bone pain in adult patients with active MFD.
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