Paradoxical SAPHO syndrome observed during anti-TNF-α therapy for Crohn's disease

Abstract

Hitoshi Amano,1 Reikei Matsuda,1 Tomohiko Shibata,2 Daisuke Takahashi,1 Shinichiro Suzuki3 1Department of Gastroenterology, Fujisawa Shonandai Hospital, Fujisawa, 2Division of Rheumatology, Department of Internal Medicine, St Marianna University School of Medicine, Yokohama City Seibu Hospital, Yokohama, 3Department of Surgery, Fujisawa Shonandai Hospital, Fujisawa, Japan Abstract: Currently, anti-TNFα antibodies are used to treat Crohn’s disease. We report on a 45-year-old Japanese female with Crohn’s disease developing SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome following exposure to the anti-TNFα antibody adalimumab. Initially, adalimumab induced remission, but the patient showed SAPHO syndrome 11 weeks following the start of adalimumab therapy for the first time. Cutaneous and articular involvement were exacerbating the condition, so adalimumab was discontinued and the patient was put on low-dose methotrexate to control her symptoms. To our knowledge, this is the first report of SAPHO syndrome occurring during anti-TNF therapy, which is thought to be a paradoxical response to adalimumab. Keywords: anti-TNFα, adalimumab, paradoxical reaction, SAPHO syndrome, Crohn’s disease

Keywords

• SAPHO syndrome
• paradoxical reaction
• Crohn disease
• anti-TNF-α therapy
• adalimumab