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SAT0036 Successful treatment of sapho syndrome with infliximab: report of two cases
  1. I Olivieri1,
  2. A Padula1,
  3. G Ciancio1,
  4. C Salvarani2,
  5. N Niccoli3,
  6. F Cantini3
  1. 1Rheumatology Department of Lucania, San Carlo Hospital, Potenza, Italy
  2. 2Rheumatic Disease Unit, Arcispedale Santa Maria Nuova, Reggio Emilia, Italy
  3. 3Rheumatic Disease Unit, Prato Hospital, Prato, Italy

Abstract

Background SAPHO syndrome (synovitis, acne, pustolosis, hyperostosis, osteitis) syndrome shares manifestations and clinical association with the spondyloarthropathy (SpA) complex.

Nonsteroidal anti-inflammatory drugs (NSAIDs) are the first choice but have limited effcacy. Second-line drugs have been tried with mixed results. Positive effects with pamidronate, which partly works by blocking tumour necrosis factor alpha (TNFalpha), have recently been reported. Infliximab, a chimeric anti-TNFalpha monoclonal IgG1 antibody, has recently been proved to be effective in ankylosing spondylitis (1) and psoriatic arthritis.1–3

Objectives To evaluate the effect of infliximab in patients with SAPHO syndrome resistant to NSAIDs and second-line drugs.

Methods Two patients meeting criteria for SAPHO syndrome and with chest wall pain limiting normal activity despite adequate treatment with NSAIDs and who had failed second-line therapy received three intravenous infusions of infliximab (5 mg/Kg) at weeks 0, 2, and 6. One of the two patients had skin manifestations consisting in severe acne at baseline. Patients were evaluated at baseline and on days 3, 7, 14 and from then every two weeks for the following variables: anterior chest wall pain, swelling and tenderness, erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP).

Results The two patients had a complete response to therapy 3 days after the first infusion of infliximab and discontinued NSAID therapy. Chest wall swelling and tenderness remitted and CRP became normal. Sever acne dramatically improved in one week after the first infusion. Patients remained symptom-free after the third infusion up to 2 months’ follow-up. Infliximab treatment was well tolerated, with no adverse event.

Conclusion Infliximab seems to be an effective drug in SAPHO syndrome. A larger, controlled, double-blind study is required.

References

  1. Brandt J, et al. Ann Rheum Dis. 2000;43:1346–52

  2. Van der Bosch F, et al. Ann Rheum Dis. 2000;59:428–33

  3. Antoni C, et al. J Rheumatol. 2000;27(Suppl 59):24

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