Ilium osteitis as the main manifestation of the SAPHO syndrome: response to infliximab therapy and review of the literature

Semin Arthritis Rheum. 2008 Apr;37(5):299-306. doi: 10.1016/j.semarthrit.2007.08.004. Epub 2007 Oct 31.

Abstract

Objective: To analyze the clinical efficacy of anti-tumor necrosis factor (TNF)-alpha 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-alpha.

Methods: A literature search of SAPHO syndrome cases treated with TNF-alpha 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-alpha therapies are effective treatment for patients with refractory SAPHO syndrome, not only for cutaneous lesions but also for persistent bone lesions such as osteitis.

Publication types

  • Review

MeSH terms

  • Acquired Hyperostosis Syndrome / complications*
  • Acquired Hyperostosis Syndrome / diagnosis
  • Acquired Hyperostosis Syndrome / drug therapy
  • Anti-Inflammatory Agents / therapeutic use*
  • Antibodies, Monoclonal / therapeutic use*
  • Diagnosis, Differential
  • Humans
  • Ilium*
  • Infliximab
  • Osteitis / diagnosis
  • Osteitis / drug therapy
  • Osteitis / etiology*
  • Treatment Outcome
  • Tumor Necrosis Factor-alpha / antagonists & inhibitors

Substances

  • Anti-Inflammatory Agents
  • Antibodies, Monoclonal
  • Tumor Necrosis Factor-alpha
  • Infliximab