title: In SAPHO syndrome anti-TNF-alpha therapy may induce persistent amelioration of osteoarticular complaints, but may exacerbate cutaneous manifestations.
abstract: OBJECTIVE
  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.affiliation: Sezione di Reumatologia, Dipartimento di Medicina Clinica e Sperimentale, Università degli Studi di Ferrara, 44100 Ferrara, Italy. alfmass@yahoo.it
authors.forename: A
authors.initials: A
authors.lastname: Massara
authors.forename: P L
authors.initials: PL
authors.lastname: Cavazzini
authors.forename: F
authors.initials: F
authors.lastname: Trotta
mesh_terms: D020083:Acquired Hyperostosis Syndrome / Q000188:drug therapy*; D000328:Adult; D000368:Aged; D000911:Antibodies, Monoclonal / Q000009:adverse effects / Q000627:therapeutic use*; D003875:Drug Eruptions / Q000209:etiology*; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D015218:Hyperostosis, Sternocostoclavicular / Q000188:drug therapy; D000069285:Infliximab; D008297:Male; D008875:Middle Aged; D010000:Osteitis / Q000188:drug therapy; D011565:Psoriasis / Q000139:chemically induced*; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors
chemical_list: D000911:Antibodies, Monoclonal; D014409:Tumor Necrosis Factor-alpha; D000069285:Infliximab