Sequential Combination Therapy Leading to Sustained Remission in a Patient with SAPHO Syndrome
C.E Huber1, §, A.G Judex1, §, J Freyschmidt2, S Feuerbach3, J Schölmerich*, 1, U Müller-Ladner1, 4
Identifiers and Pagination:Year: 2009
First Page: 18
Last Page: 21
Publisher Id: TORJ-3-18
Article History:Received Date: 30/12/2008
Revision Received Date: 14/1/2009
Acceptance Date: 25/2/2009
Electronic publication date: 27/3/2009
Collection year: 2009
open-access license: This is an open access article licensed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/)which permits unrestricted, non-commercial use, distribution and reproduction in any medium, provided the work is properly cited.
The SAPHO syndrome represents a variety of clinically similar disorders with the key features of hyperostotic bone lesions in combination with chronic pustular skin disease. The respective pathophysiology of bone and joint manifestations in SAPHO syndrome is still a matter of discussion. For example it does not appear to represent reactive arthritis and HLA B27 antigen, with the latter being typically present in patients with spondyloarthopathies. Treatment of SAPHO syndrome is also not well established and consists of various antiinflammatory and antirheumatic drugs. Here, we report a female patient with active SAPHO syndrome suffering from sternal swelling of unknown origin that had been known for 10 years and a 4-year-history of severe lower back pain. Remarkable were also a typical pustulous palmar erythema associated with swelling and decreased motility of both MCP-I joints. Inflammation parameters were high with an ESR 68 mm/1st hour and a CRP of 19.6 mg/l. She was initially treated with rofecoxib and doxycycline, followed by sulfasalazine with only partial clinical response. Thereafter, both articular symptoms as well as cutaneous lesions responded well to a combination therapy with methotrexate and sulfasalazine. Thus, the case illustrates nicely that methotrexate in combination with another DMARD can be successfully applied to patients with long-term active SAPHO syndrome.