Regression of fibrous dysplasia in response to denosumab therapy: A report of two cases
Maartje E. Meier,
Wouter van der Bruggen,
Michiel A.J. van de Sande,
Natasha M. Appelman-Dijkstra
Affiliations
Maartje E. Meier
Department of Orthopedic Surgery, Center for Bone Quality, Leiden University Medical Center (LUMC), Albinusdreef 2, 2333ZA Leiden, the Netherlands; Corresponding author.
Wouter van der Bruggen
Section of Nuclear Medicine, Department of Radiology, LUMC, Albinusdreef 2, 2333ZA Leiden, the Netherlands; Department of Nuclear Medicine, Slingeland Hospital, Kruisbergseweg 25, 7009 BL Doetinchem, the Netherlands
Michiel A.J. van de Sande
Department of Orthopedic Surgery, Center for Bone Quality, Leiden University Medical Center (LUMC), Albinusdreef 2, 2333ZA Leiden, the Netherlands
Natasha M. Appelman-Dijkstra
Department of Internal Medicine, Division of Endocrinology, Center for Bone Quality, LUMC, Albinusdreef 2, 2333ZA Leiden, the Netherlands
We present two patients with fibrous dysplasia who showed a decrease in lesional size and activity after denosumab therapy. Both patients also experienced a reduction in pain and bone turnover markers, which had not been accomplished during previous bisphosphonate therapy. These cases highlight the potential of denosumab to decrease lesional size in fibrous dysplasia. This finding has been reported in mice, but not in humans. Denosumab may be considered when bisphosphonates are not tolerated or not effective (enough), or in severe cases as neoadjuvant therapy to improve surgical possibilities and outcome. In addition, these results show that Na[18F]F PET-CT is suitable for detecting change in each fibrous dysplasia lesion distinctively.
