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. 2023 Feb 23;388(8):766-768.
doi: 10.1056/NEJMc2214862.

Safety and Efficacy of Denosumab for Fibrous Dysplasia of Bone

Luis F de Castro  1 , Zachary Michel  1 , Kristen Pan  1 , Jocelyn Taylor  1 , Vivian Szymczuk  1 , Sriram Paravastu  1 , Babak Saboury  1 , Georgios Z Papadakis  2 , Xiaobai Li  1 , Kelly Milligan  1 , Brendan Boyce  3 , Scott M Paul  4 , Michael T Collins  4 , Alison M Boyce  4
Affiliations

Safety and Efficacy of Denosumab for Fibrous Dysplasia of Bone

Luis F de Castro et al. N Engl J Med. .
No abstract available

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Figures

Figure 1.
Figure 1.. Effect of Denosumab Treatment on Bone-Turnover Markers and Lesion Activity as Assessed Radiologically.
Panel A shows the changes in the levels of the bone-formation marker procollagen type 1 N-terminal propeptide (P1NP) and the levels of the bone-resorption marker C-terminal telopeptide (CTX) in each participant. The dashed lines indicate the normal range for each assay. Panel B shows representative images from positron-emission tomography–computed tomography (PET–CT), which was performed after the administration of 18F–sodium fluoride tracer. Qualitative changes in tracer uptake by the fibrous dysplasia lesions are shown in images from Participants DB01 (top), DB02 (bottom left), and DB09 (bottom right). Panel C shows representative stains of bone-biopsy specimens before (baseline) and 6 months after treatment with denosumab. Hematoxylin and eosin staining (images on left) shows increased lesional bone formation (Participant DB04). Immunohistochemical staining (images in middle) shows decreased expression of the cell-proliferation marker minichromosome maintenance 2 (MCM2) (Participant DB02). In situ hybridization staining (images on right) shows upregulation in mRNA expression of the bone-formation marker sclerostin (encoded by SOST) (Participant DB07). The dashed boxes refer to the insets. Panel D shows the effects of discontinuation of denosumab treatment on bone-turnover markers. The graphs show the percent changes from baseline in the resorption marker CTX (upper graph) and in the formation marker P1NP (lower graph) in each participant. The Greek letter Ω corresponds to the occurrence of severe hypercalcemia, and δ corresponds to the occurrence of mild asymptomatic hypercalcemia.
See this image and copyright information in PMC

References

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    1. de Castro LF, Burke AB, Wang HD, et al. Activation of RANK/RANKL/OPG pathway is involved in the pathophysiology of fibrous dysplasia and associated with disease burden. J Bone Miner Res 2019; 34: 290–4. - PMC - PubMed
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    1. van der Bruggen W, Vriens D, Meier ME, et al. Denosumab reduces lesional fluoride skeletal burden on Na[18F]F PET-CT in patients with fibrous dysplasia/McCune-Albright syndrome. J Clin Endocrinol Metab 2021; 106(8): e2980–e2994. - PMC - PubMed
    1. Papadakis GZ, Manikis GC, Karantanas AH, et al. 18F-NaF PET/CT imaging in fibrous dysplasia of bone. J Bone Miner Res 2019; 34: 1619–31. - PMC - PubMed

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