title: The SAPHO syndrome: a single-center study of 41 adult patients.
abstract: OBJECTIVE
  The SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis, osteitis) is a rare disease and only a few series have been published about it. We describe the experience of a rheumatology department with a focus on the therapeutic aspects.
  
  
  METHODS
  All cases of SAPHO fulfilling the Benhamou criteria and seen between 1992 and 2013 in our unit were retrospectively included.
  
  
  RESULTS
  Forty-one patients (11 men and 30 women) were included. The average age was 45 years (20-80 yrs) at the time of diagnosis. Nineteen patients had palmoplantar pustulosis, 3 had psoriasis vulgaris, 3 had severe acne, and 15 had no dermatological involvement. Bone symptoms involved mainly the anterior chest wall (n = 28, 68%), followed by the spine (n = 16, 39%) and sacroiliac joints (n = 12, 29%). Four patients had peripheral osteitis and 2 had mandibular osteitis. Ten patients had peripheral arthritis affecting mainly the knees and wrists. None of the 36 patients tested was HLA-B27-positive. At least a partial response was achieved with colchicine, methotrexate, or sulfasalazine in 0/6, 2/4, and 1/6 of patients, respectively. Antibiotic therapy (azithromycin, n = 7; doxycycline, n = 2) was effective in 2/9 patients. Pamidronate was prescribed in 26 patients with bone involvement and 18/22 patients evaluable at 6 months responded to this therapy. Two patients were treated with tumor necrosis factor blockers: 1 with infliximab and 1 with adalimumab, followed by etanercept.
  
  
  CONCLUSIONS
  Based on our experience, SAPHO can be diagnosed in the elderly. It was not associated with HLA-B27. Soft tissue involvement was common and the bisphosphonates were generally effective on bone involvement.
authors.affiliation: From the Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France.F. Aljuhani, MD; A. Tournadre, MD; Z. Tatar, MD; M. Couderc, MD; S. Mathieu, MD; S. Malochet-Guinamand, MD; M. Soubrier, MD, PhD; J-J. Dubost, MD; Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand.
authors.forename: Faisal
authors.initials: F
authors.lastname: Aljuhani
authors.affiliation: From the Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France.F. Aljuhani, MD; A. Tournadre, MD; Z. Tatar, MD; M. Couderc, MD; S. Mathieu, MD; S. Malochet-Guinamand, MD; M. Soubrier, MD, PhD; J-J. Dubost, MD; Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand.
authors.forename: Anne
authors.initials: A
authors.lastname: Tournadre
authors.affiliation: From the Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France.F. Aljuhani, MD; A. Tournadre, MD; Z. Tatar, MD; M. Couderc, MD; S. Mathieu, MD; S. Malochet-Guinamand, MD; M. Soubrier, MD, PhD; J-J. Dubost, MD; Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand.
authors.forename: Zuzana
authors.initials: Z
authors.lastname: Tatar
authors.affiliation: From the Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France.F. Aljuhani, MD; A. Tournadre, MD; Z. Tatar, MD; M. Couderc, MD; S. Mathieu, MD; S. Malochet-Guinamand, MD; M. Soubrier, MD, PhD; J-J. Dubost, MD; Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand.
authors.forename: Marion
authors.initials: M
authors.lastname: Couderc
authors.affiliation: From the Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France.F. Aljuhani, MD; A. Tournadre, MD; Z. Tatar, MD; M. Couderc, MD; S. Mathieu, MD; S. Malochet-Guinamand, MD; M. Soubrier, MD, PhD; J-J. Dubost, MD; Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand.
authors.forename: Sylvain
authors.initials: S
authors.lastname: Mathieu
authors.affiliation: From the Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France.F. Aljuhani, MD; A. Tournadre, MD; Z. Tatar, MD; M. Couderc, MD; S. Mathieu, MD; S. Malochet-Guinamand, MD; M. Soubrier, MD, PhD; J-J. Dubost, MD; Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand.
authors.forename: Sandrine
authors.initials: S
authors.lastname: Malochet-Guinamand
authors.affiliation: From the Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France.F. Aljuhani, MD; A. Tournadre, MD; Z. Tatar, MD; M. Couderc, MD; S. Mathieu, MD; S. Malochet-Guinamand, MD; M. Soubrier, MD, PhD; J-J. Dubost, MD; Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand.
authors.forename: Martin
authors.initials: M
authors.lastname: Soubrier
authors.affiliation: From the Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand, Clermont-Ferrand, France.F. Aljuhani, MD; A. Tournadre, MD; Z. Tatar, MD; M. Couderc, MD; S. Mathieu, MD; S. Malochet-Guinamand, MD; M. Soubrier, MD, PhD; J-J. Dubost, MD; Department of Rheumatology, Gabriel Montpied Hospital, Centre Hospitalier Universitaire de Clermont-Ferrand. jjdubost@chu-clermontferrand.fr.
authors.forename: Jean-Jacques
authors.initials: JJ
authors.lastname: Dubost
mesh_terms: D020083:Acquired Hyperostosis Syndrome / Q000150:complications / Q000175:diagnosis*; D000328:Adult; D000368:Aged; D000369:Aged, 80 and over; D001168:Arthritis / Q000150:complications*; D005260:Female; D006801:Humans; D008297:Male; D008875:Middle Aged; D012189:Retrospective Studies; D055815:Young Adult
keywords: BISPHOSPHONATE; PALMOPLANTAR PUSTULOSIS; SAPHO; SPONDYLOARTHRITIS