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Long-Term Treatment of Severe SAPHO Syndrome with Adalimumab

Case Report and a Review of the Literature

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Abstract

SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome defines an association of inflammatory cutaneous disorders with osteoarticular manifestations and represents a clinical and therapeutic challenge. We report a case of severe SAPHO syndrome with acne conglobata and a diffuse involvement of the anterior chest wall and sacroiliac joints that required treatment with isotretinoin and adalimumab, a new fully human anti-tumor necrosis factor (TNF)-α monoclonal antibody. Combination treatment determined a complete clinical remission of cutaneous and osteoarticular manifestations after 48 weeks. Despite maintenance of clinical remission, follow-up imaging studies after 24 months of adalimumab monotherapy revealed osteoarticular disease progression, with features of inflammatory osteitis.

TNFα antagonists have been used as third-line therapy for SAPHO syndrome in single case reports or case series, but these lack consistent long-term follow-up. SAPHO syndrome can present an intermittentfavorable course in the majority of cases as well as a chronic-progressive course, the latter requiring aggressive combination treatment with TNFα antagonists and conventional systemic agents.

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Acknowledgments

No sources of funding were used to prepare this case report. The authors have no conflicts of interest that are directly relevant to the content of this case report.

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Correspondence to Simone Garcovich.

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Garcovich, S., Amelia, R., Magarelli, N. et al. Long-Term Treatment of Severe SAPHO Syndrome with Adalimumab. Am J Clin Dermatol 13, 55–59 (2012). https://doi.org/10.2165/11593250-000000000-00000

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