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Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[(18)F]F PET-CT in Patients With Fibrous Dysplasia/McCune–Albright Syndrome
CONTEXT: The correlation between fibrous dysplasia/McCune–Albright syndrome (FD/MAS) skeletal disease burden on Na[(18)F]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 F...
Autores principales: | , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Oxford University Press
2021
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8277209/ https://www.ncbi.nlm.nih.gov/pubmed/33788944 http://dx.doi.org/10.1210/clinem/dgab212 |
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author | van der Bruggen, Wouter Vriens, Dennis Meier, Maartje E Smit, Frits Winter, Elizabeth M de Geus-Oei, Lioe-Fee Appelman-Dijkstra, Natasha M |
author_facet | van der Bruggen, Wouter Vriens, Dennis Meier, Maartje E Smit, Frits Winter, Elizabeth M de Geus-Oei, Lioe-Fee Appelman-Dijkstra, Natasha M |
author_sort | van der Bruggen, Wouter |
collection | PubMed |
description | CONTEXT: The correlation between fibrous dysplasia/McCune–Albright syndrome (FD/MAS) skeletal disease burden on Na[(18)F]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. OBJECTIVE: To investigate treatment response measurements in patients with FD/MAS who underwent Na[(18)F]F-PET-CT and treatment with antiresorptives. METHODS: 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[(18)F]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[(18)F]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. RESULTS: Disease activity decreased significantly, with FTV 361 cm(3) to 97 cm(3) (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). CONCLUSION: Na[(18)F]F-PET-CT captured treatment-induced lesional changes which correlated with BTMs and pain reduction. Therefore Na[(18)F]F-PET-CT can be used as an objective local parameter of response to denosumab treatment in FD/MAS. |
format | Online Article Text |
id | pubmed-8277209 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Oxford University Press |
record_format | MEDLINE/PubMed |
spelling | pubmed-82772092021-07-15 Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[(18)F]F PET-CT in Patients With Fibrous Dysplasia/McCune–Albright Syndrome van der Bruggen, Wouter Vriens, Dennis Meier, Maartje E Smit, Frits Winter, Elizabeth M de Geus-Oei, Lioe-Fee Appelman-Dijkstra, Natasha M J Clin Endocrinol Metab Online Only Articles CONTEXT: The correlation between fibrous dysplasia/McCune–Albright syndrome (FD/MAS) skeletal disease burden on Na[(18)F]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. OBJECTIVE: To investigate treatment response measurements in patients with FD/MAS who underwent Na[(18)F]F-PET-CT and treatment with antiresorptives. METHODS: 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[(18)F]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[(18)F]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. RESULTS: Disease activity decreased significantly, with FTV 361 cm(3) to 97 cm(3) (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). CONCLUSION: Na[(18)F]F-PET-CT captured treatment-induced lesional changes which correlated with BTMs and pain reduction. Therefore Na[(18)F]F-PET-CT can be used as an objective local parameter of response to denosumab treatment in FD/MAS. Oxford University Press 2021-03-31 /pmc/articles/PMC8277209/ /pubmed/33788944 http://dx.doi.org/10.1210/clinem/dgab212 Text en © The Author(s) 2021. Published by Oxford University Press on behalf of the Endocrine Society. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) ), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com |
spellingShingle | Online Only Articles van der Bruggen, Wouter Vriens, Dennis Meier, Maartje E Smit, Frits Winter, Elizabeth M de Geus-Oei, Lioe-Fee Appelman-Dijkstra, Natasha M Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[(18)F]F PET-CT in Patients With Fibrous Dysplasia/McCune–Albright Syndrome |
title | Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[(18)F]F PET-CT in Patients With Fibrous Dysplasia/McCune–Albright Syndrome |
title_full | Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[(18)F]F PET-CT in Patients With Fibrous Dysplasia/McCune–Albright Syndrome |
title_fullStr | Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[(18)F]F PET-CT in Patients With Fibrous Dysplasia/McCune–Albright Syndrome |
title_full_unstemmed | Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[(18)F]F PET-CT in Patients With Fibrous Dysplasia/McCune–Albright Syndrome |
title_short | Denosumab Reduces Lesional Fluoride Skeletal Burden on Na[(18)F]F PET-CT in Patients With Fibrous Dysplasia/McCune–Albright Syndrome |
title_sort | denosumab reduces lesional fluoride skeletal burden on na[(18)f]f pet-ct in patients with fibrous dysplasia/mccune–albright syndrome |
topic | Online Only Articles |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8277209/ https://www.ncbi.nlm.nih.gov/pubmed/33788944 http://dx.doi.org/10.1210/clinem/dgab212 |
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