TY - JOUR
T1 - Pathophysiology and medical treatment of pain in fibrous dysplasia of bone
AU - Chapurlat, Roland D.
AU - Gensburger, Deborah
AU - Jimenez-Andrade, Juan M.
AU - Ghilardi, Joseph R.
AU - Kelly, Marilyn
AU - Mantyh, Patrick
N1 - Funding Information:
This article has been published as part of Orphanet Journal of Rare Diseases Volume 7 Supplement 1, 2012: International Meeting on Fibrous Dysplasia/ McCune-Albright Syndrome and Cherubism. The full contents of the supplement are available online at http://www.ojrd.com/supplements/7/S1. Publication of the proceedings was funded by the Fibrous Dysplasia Foundation and an unrestricted grant from Zimmer.
PY - 2012
Y1 - 2012
N2 - 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.
AB - 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.
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U2 - 10.1186/1750-1172-7-S1-S3
DO - 10.1186/1750-1172-7-S1-S3
M3 - Article
C2 - 22640953
AN - SCOPUS:84861374467
SN - 1750-1172
VL - 7
JO - Orphanet Journal of Rare Diseases
JF - Orphanet Journal of Rare Diseases
IS - SUPPL. 1
M1 - S3
ER -