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.