In SAPHO syndrome anti-TNF-alpha therapy may induce persistent amelioration of osteoarticular complaints, but may exacerbate cutaneous manifestations.

Abstract

OBJECTIVES:

SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis and osteitis) is a rare disease combining skin, bone and joint manifestations. In recent years new therapeutic strategies have been tried, among them TNF-alpha-blocking agents. We report our experience with infliximab in four cases of SAPHO syndrome refractory to conventional therapies.

METHODS:

Between 2002 and 2005, four cases of SAPHO syndrome (two females and two males; mean age 49.7 yr) responding poorly to conventional drugs were treated with infliximab. The dose was 5 mg/kg, according to the protocol used in spondyloarthropathies, with infusions at 0, 2 and 6 weeks followed by 6 weeks intervals. No active cutaneous manifestations were present at the time of starting therapy.

RESULTS:

Complete remission of osteoarticular involvement was achieved after the second or third infusion, and the positive response was maintained for up to 12 months. A patient relapsed after discontinuation of infliximab, because of infectious complication. Palmoplantaris pustulosis relapsed in two patients after three and six infusions, respectively; there was slight improvement after discontinuation of anti-TNF-alpha drugs.

CONCLUSIONS:

Infliximab seems to be a very effective therapy for osteoarticular complaints of SAPHO syndrome. Cutaneous involvement responded less favourably, palmoplantaris pustulosis relapse being a possible complication.

• Authors: A Massara, P L Cavazzini, F Trotta
• Journal: Rheumatology (Oxford, England) (Rheumatology (Oxford)) Vol. 45 Issue 6 Pg. 730-3 (Jun 2006) ISSN: 1462-0324 [Print] England
• PMID: 16403830 (Publication Type: Case Reports, Journal Article)

Chemical References

• Antibodies, Monoclonal
• Tumor Necrosis Factor-alpha
• Infliximab

Topics

• Acquired Hyperostosis Syndrome (drug therapy)
• Adult
• Aged
• Antibodies, Monoclonal (adverse effects, therapeutic use)
• Drug Eruptions (etiology)
• Female
• Follow-Up Studies
• Humans
• Hyperostosis, Sternocostoclavicular (drug therapy)
• Infliximab
• Male
• Middle Aged
• Osteitis (drug therapy)
• Psoriasis (chemically induced)
• Tumor Necrosis Factor-alpha (antagonists & inhibitors)