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Published 20 May 2009
Cite this as: BMJ Case Reports 2009 [doi:10.1136/bcr.10.2008.1145]
Copyright © 2009 by the BMJ Publishing Group Ltd.

Unexpected outcome (positive or negative) including adverse drug reactions

Response to infliximab in SAPHO syndrome

Julia Fruehauf1, Brigitte Cierny-Modrè1, Laila El-Shabrawi Caelen1, Thomas Schwarz2, Roland Weinke2, Elisabeth Aberer1

1 Medical University of Graz, Department of Dermatology, Auenbruggerplatz 8, Graz, Graz, 8036, Austria
2 Medical University of Graz, Department of Radiology, Auenbruggerplatz 9, Graz, 8036, Austria

Correspondence to:
Julia Fruehauf, JuliaFruehauf{at}mac.com

SUMMARY

Infliximab has become increasingly important in the treatment of SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome. There is, however, little experience with this biological agent, and treatment protocols usually follow the regimens for spondylarthropathies. We report a patient with a highly unusual and severe clinical presentation of SAPHO syndrome including widespread bone and skin disease, and collagenous colitis. Infliximab treatment (5 mg/kg) given at weeks 0, 2 and 6 and every 8 weeks thereafter, induced rapid remission of the osteoarticular symptoms, although the skin lesions improved only partially, and after 10 months continuous therapy with infliximab a bone scan even uncovered new active bone lesions. Collagenous colitis is unresponsive to tumour necrosis factor {alpha} (TNF{alpha}) blocking agents. This moderate response to infliximab may indicate that a more aggressive treatment protocol is mandatory. We further believe that remission of osteoarticular complaints should be routinely confirmed by scintigraphic findings to verify treatment response.


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