{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T06:23:29Z","timestamp":1793255009409,"version":"4.5.0"},"reference-count":18,"publisher":"The Endocrine Society","issue":"8","license":[{"start":{"date-parts":[[2021,3,31]],"date-time":"2021-03-31T00:00:00Z","timestamp":1617148800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,7,13]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>The correlation between fibrous dysplasia\/McCune\u2013Albright syndrome (FD\/MAS) skeletal disease burden on Na[18F]F positron emission tomography\u2013computed 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.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>To investigate treatment response measurements in patients with FD\/MAS who underwent Na[18F]F-PET-CT and treatment with antiresorptives.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>Observational 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\u2005=\u20058) treatment and non-denosumab patients (n\u2005=\u20057). On Na[18F]F-PET-CT the volumetric measures of FD burden (fluoride tumor volume [FTV]) and \u201cfraction affected skeleton\u201d (FAS) represented the portion of the skeleton affected. This was correlated with BTMs and pain.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Disease activity decreased significantly, with FTV 361 cm3 to 97 cm3 (P\u2005=\u2005.018) in denosumab-treated patients, but not in non-denosumab patients (P\u2005=\u2005.249). Serum P1NP and alkaline phosphatase (ALP) decreased significantly: 82 ng\/mL vs 55 ng\/mL (P\u2005=\u2005.023) and 119 IU\/L vs 84 IU\/L (P\u2005=\u2005.020), 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\u2005=\u20050.730, P\u2005&amp;lt;\u2005.001; and ALP r\u2005=\u20050.406, P\u2005=\u2005.006), as did change in BTMs and FTV: P1NP (P\u2005=\u20050.032) and ALP (P\u2005=\u20050.024). FAS strongly correlated with treatment-induced decrease in ALP (P\u2005=\u2005.027) and P1NP (P\u2005=\u2005.009).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusion<\/jats:title>\n                  <jats:p>Na[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.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgab212","type":"journal-article","created":{"date-parts":[[2021,3,31]],"date-time":"2021-03-31T18:16:51Z","timestamp":1617214611000},"page":"e2980-e2994","source":"Crossref","is-referenced-by-count":27,"title":["Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[18F]F PET-CT in Patients With Fibrous Dysplasia\/McCune\u2013Albright Syndrome"],"prefix":"10.1210","volume":"106","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4689-6506","authenticated-orcid":false,"given":"Wouter","family":"van der Bruggen","sequence":"first","affiliation":[{"name":"Section of Nuclear Medicine, Department of Radiology, Leiden University Medical Center (LUMC), Leiden, The Netherlands"},{"name":"Department of Nuclear Medicine, Slingeland Hospital, Doetinchem, The 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Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth M","family":"Winter","sequence":"additional","affiliation":[{"name":"Center for Bone Quality, Dept. of Internal Medicine, Division of Endocrinology, Leiden University Medical Center (LUMC), Leiden, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lioe-Fee","family":"de Geus-Oei","sequence":"additional","affiliation":[{"name":"Section of Nuclear Medicine, Department of Radiology, Leiden University Medical Center (LUMC), Leiden, The Netherlands"},{"name":"Biomedical Photonic Imaging Group, University of Twente, Enschede, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natasha M","family":"Appelman-Dijkstra","sequence":"additional","affiliation":[{"name":"Center for Bone Quality, Dept. of Internal Medicine, Division of Endocrinology, Leiden University Medical Center (LUMC), Leiden, The 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