Clinical improvement in a patient with monostotic melorheostosis after treatment with denosumab: a case report

TY - JOUR

T1 - Clinical improvement in a patient with monostotic melorheostosis after treatment with denosumab

T2 - a case report

AU - Byberg, Sarah

AU - Abrahamsen, Bo

AU - Kassem, Moustapha

AU - Ralston, Stuart

AU - Schwarz, Peter

PY - 2018/9/27

Y1 - 2018/9/27

N2 - BACKGROUND: A 20-year-old Danish woman with melorheostosis in her right femoral shaft and disabling pain in the affected area, whose symptoms did not in the long term respond to zoledronic acid, experienced continuous remission of pain after treatment with denosumab. To the best of our knowledge, this is the first case report on denosumab treatment for melorheostosis.CASE PRESENTATION: Radiologic findings and bone biopsy showed irregular cortical hyperostosis in the right femoral shaft with increased tracer uptake on Tc99-bone scan. The diagnosis of melorheostosis was made based on the radiological findings. There was a good initial response to zoledronic acid administration, but after relapse of pain, the second and third administrations had a poor effect. As a second line of treatment denosumab was administered at 8-week intervals, the frequency was based on our patient's symptoms and on biochemical markers of bone turnover.CONCLUSION: This is the first report indicating that denosumab has a place in the treatment of melorheostosis when the effect of bisphosphonate treatment is insufficient.

AB - BACKGROUND: A 20-year-old Danish woman with melorheostosis in her right femoral shaft and disabling pain in the affected area, whose symptoms did not in the long term respond to zoledronic acid, experienced continuous remission of pain after treatment with denosumab. To the best of our knowledge, this is the first case report on denosumab treatment for melorheostosis.CASE PRESENTATION: Radiologic findings and bone biopsy showed irregular cortical hyperostosis in the right femoral shaft with increased tracer uptake on Tc99-bone scan. The diagnosis of melorheostosis was made based on the radiological findings. There was a good initial response to zoledronic acid administration, but after relapse of pain, the second and third administrations had a poor effect. As a second line of treatment denosumab was administered at 8-week intervals, the frequency was based on our patient's symptoms and on biochemical markers of bone turnover.CONCLUSION: This is the first report indicating that denosumab has a place in the treatment of melorheostosis when the effect of bisphosphonate treatment is insufficient.

U2 - 10.1186/s13256-018-1820-y

DO - 10.1186/s13256-018-1820-y

M3 - Journal article

C2 - 30257703

SN - 1752-1947

VL - 12

SP - 1

EP - 6

JO - Journal of Medical Case Reports

JF - Journal of Medical Case Reports

M1 - 278

ER -