Ilium osteitis as the main manifestation of the SAPHO syndrome: response to infliximab therapy and review of the literature.
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.
Moll
Concepción
C
Arthritis Unit, Rheumatology Department, Hospital Clínic, University of Barcelona, Barcelona, Spain.
Hernández
M Victoria
MV
Cañete
Juan D
JD
Gómez-Puerta
José A
JA
Soriano
Alex
A
Collado
Antonio
A
Sanmartí
Raimon
R
D020083:Acquired Hyperostosis Syndrome / Q000150:complications* / Q000175:diagnosis / Q000188:drug therapy; D000893:Anti-Inflammatory Agents / Q000627:therapeutic use*; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D003937:Diagnosis, Differential; D006801:Humans; D007085:Ilium; D000069285:Infliximab; D010000:Osteitis / Q000175:diagnosis / Q000188:drug therapy / Q000209:etiology*; D016896:Treatment Outcome; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors
D000893:Anti-Inflammatory Agents; D000911:Antibodies, Monoclonal; D014409:Tumor Necrosis Factor-alpha; D000069285:Infliximab