This article is part of the supplement: International Meeting on Fibrous Dysplasia/McCune-Albright Syndrome and Cherubism

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Pathophysiology and medical treatment of pain in fibrous dysplasia of bone

Roland D Chapurlat12*, Deborah Gensburger12, Juan M Jimenez-Andrade3, Joseph R Ghilardi4, Marilyn Kelly5 and Patrick Mantyh346

  • * Corresponding author: Roland D Chapurlat

Author Affiliations

1 INSERM UMR 1033, Université de Lyon, Hospices Civils de Lyon, Hôpital E Herriot, 69437 Lyon, France

2 National Reference Center for Fibrous Dysplasia of Bone, Hôpital E Herriot, 69437 Lyon, France

3 Department of Pharmacology, College of Medicine, University of Arizona, Tucson, AZ 85724, USA

4 Research Service, VA Medical Center, Minneapolis, MN 55417, USA

5 Skeletal Clinical Studies Unit, Craniofacial and Skeletal Diseases Branch, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, MD, USA

6 Arizona Cancer Center, University of Arizona, Tucson, AZ 85724, USA

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Orphanet Journal of Rare Diseases 2012, 7(Suppl 1):S3  doi:10.1186/1750-1172-7-S1-S3

Published: 24 May 2012


One of the most common complications of fibrous dysplasia of bone (FD) is bone pain. Usual pain killers are often of inadequate efficacy to control this bone pain. The mechanism of bone pain in FD remains uncertain, but by analogy with bone tumors one may consider that ectopic sprouting and formation of neuroma-like structures by sensory and sympathetic nerve fibers also occur in the dysplastic skeleton. Bone pain has been reported in up to 81% of adults and 49% of children. It affects predominantly the lower limbs and the spine. The degree of pain is highly variable and adults reports more pain than children. Bisphosphonates have been shown to reduce bone pain in uncontrolled studies. Their influence on bone strength remains unknown. In a randomized trial testing alendronate, bone pain was not significantly improved. Another trial assessing the effect of risedronate is ongoing. Possible future therapies include tocilizumab, denosumab and drugs targeting nerve growth factor and its receptor TrkA.