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
1 Department of Internal Medicine I, University of Regensburg, Regensburg, Germany
2 Department of Radiology, Zentralkrankenhaus St.-Jürgen-Strasse, Bremen, Germany
3 Department of Radiology, University of Regensburg, Regensburg, Germany
4 Department of Internal Medicine and Rheumatology, Justus-Liebig University Giessen/Kerckhoff Clinic Bad Nauheim, Germany

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© Huber et al.; Licensee Bentham Open.

open-access license: This is an open access article licensed under the terms of the Creative Commons Attribution Non-Commercial License ( permits unrestricted, non-commercial use, distribution and reproduction in any medium, provided the work is properly cited.

* Address correspondence to this author at the Department of Internal Medicine I, University of Regensburg Medical Center, Franz-Josef-Strauss Allee 11, D-93053 Regensburg, Germany; Tel: +49 941 944 7001; Fax: +49 941944 7002; E-mail:
§ These authors contributed equally to this paper.


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.

Keywords: Case report, combination therapy, doxycycline, methotrexate, rofecoxib, SAPHO, sulfasalazine, treatment..