RT Journal Article
SR Electronic
T1 Tumor Necrosis Factor-α Blockers in SAPHO Syndrome
JF The Journal of Rheumatology
JO J Rheumatol
FD The Journal of Rheumatology
SP jrheum.091086
DO 10.3899/jrheum.091086
A1 Kaouther Ben Abdelghani
A1 Delphine Gerard Dran
A1 Jacques-Eric Gottenberg
A1 Jacques Morel
A1 Jean Sibilia
A1 Bernard Combe
YR 2010
UL http://www.jrheum.org/content/early/2010/05/12/jrheum.091086.abstract
AB Objective To analyze the clinical efficacy of anti-tumor necrosis factor-α (TNF-α) therapy in treatment of synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome, we describe cases of refractory SAPHO syndrome and review cases treated with anti-TNF-α reported in the literature. Methods We describe 6 cases of patients with SAPHO syndrome treated with anti-TNF-α between 2004 and 2008. Therapeutic response was evaluated according to improvement in pain score, amelioration of disease activity, and improvement in function. The efficacy of treatment was considered to be reduced need for analgesics and/or antiinflammatory therapy. Results In our series, 4 patients received infliximab, 1 etanercept, and 1 adalimumab. These treatments brought clinical response in 4 patients (66.6%): response was sustained with infliximab in 1 case for 7 months; with adalimumab in another case for 22 months; and with etanercept in 2 cases for 1 and 42 months, respectively. In contrast, 2 other patients showed no response to infliximab. Improvement was initially temporary after infusions 1 and 2, then pain recurred at Week 14. Skin lesions were healed in 3 of 4 cases, but recurred or worsened in 2 cases, after infusion 2 of infliximab. Treatment was generally well tolerated. Paradoxical psoriasis was noted in 2 cases and urticaria in 1. Conclusion Given our results and those from the literature, TNF-α blockers should be considered in the therapeutic strategy of refractory cases of SAPHO syndrome, despite their effect seeming less impressive than in other spondyloarthropathies.