Ilium Osteitis as the Main Manifestation of the SAPHO Syndrome: Response to Infliximab Therapy and Review of the Literature

• Published in: Seminars in arthritis and rheumatism Vol. 37; no. 5; pp. 299 - 306
• Main Authors: Moll, Concepción, MD, Hernández, M. Victoria, MD, Cañete, Juan D., MD, PhD, Gómez-Puerta, José A., MD, Soriano, Alex, MD, Collado, Antonio, MD, Sanmartí, Raimon, MD
• Format: Journal Article
• Language: English
• Published: Philadelphia, PA Elsevier Inc 01.04.2008; Elsevier
• Subjects: Acquired Hyperostosis Syndrome - complications; Acquired Hyperostosis Syndrome - diagnosis; Acquired Hyperostosis Syndrome - drug therapy; Anti-Inflammatory Agents - therapeutic use; Antibodies, Monoclonal - therapeutic use; Bacterial arthritis and osteitis; Bacterial diseases; Biological and medical sciences; Bullous diseases of the skin; Dermatology; Diagnosis, Differential; Diseases of the osteoarticular system; Human bacterial diseases; Humans; Ilium; Ilium osteitis; Infectious diseases; Infliximab; Medical sciences; Osteitis - diagnosis; Osteitis - drug therapy; Osteitis - etiology; Palmoplantar pustulosis; Rheumatology; SAPHO syndrome; Treatment Outcome; Tumor necrosis factor alpha blockers; Tumor Necrosis Factor-alpha - antagonists & inhibitors; Ilium osteitis; Infliximab; Tumor necrosis factor alpha blockers; SAPHO syndrome; Palmoplantar pustulosis; Palmoplantaris pustulosis; Skin disease; Pustulosis dermatosis; Cytokine; Diseases of the osteoarticular system; Rheumatology; Monoclonal antibody; Review; Anti-Tumor Necrosis Factor-alpha; Immunomodulator; Treatment; Osteitis; Anticytokine; Antirheumatic agent; Immunosuppressive agent; Tumor necrosis factor α; Bibliographic review
• ISSN: 0049-0172; 1532-866X
• DOI: 10.1016/j.semarthrit.2007.08.004

Objective To analyze the clinical efficacy of anti-tumor necrosis factor (TNF)-α therapy in the SAPHO (synovitis, acne, pustulosis, hyperostosis, osteitis) syndrome. We describe 2 new cases with ilium osteitis as the main SAPHO syndrome feature and review reported cases treated with anti-TNF-α. Methods A literature search of SAPHO syndrome cases treated with TNF-α blocking therapy with special emphasis on osteoarticular and skin responses was performed. Results Eighteen cases were identified: 17 SAPHO syndrome and 1 chronic recurrent multifocal osteomyelitis, a juvenile variant of SAPHO syndrome. Sixteen were reported cases and 2 were nonreported cases seen in our arthritis unit. Sixteen patients received infliximab and 2 received etanercept, with an early, sustained clinical improvement in most cases. Conclusions Anti-TNF-α therapies are effective treatment for patients with refractory SAPHO syndrome, not only for cutaneous lesions but also for persistent bone lesions such as osteitis.