title: SAPHO: Treatment options including bisphosphonates.
abstract: Both the diagnosis and treatment of the syndrome of synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) remain difficult. We describe a case series of 21 patients with SAPHO and their response to several pharmacological treatments.
  
  
  
  Clinical and biochemical data, along with medical imaging, were collected from the medical records of 21 patients, diagnosed as SAPHO during follow-up between 2005 and 2013. Symptoms and inflammatory markers were recorded twice, once at first patient presentation, and once at the end of follow-up. Synovitis, acne, pustulosis, hyperostosis, and osteitis were labeled as defining features. All treatment options were categorized according to their respective responses (full remission, partial remission, and no disease control).
  
  
  
  There was a female predominance and a median age of 32 years (range: 12-54 years). Median follow-up duration was 45 months (range: 0-188 months). Total prevalence of defining features in this cohort increased for each defining feature during follow-up, except for acne. All patients reached full or partial remission at the end of follow-up. A total of 14 patients were treated with bisphosphonates. Of which 8 of them went into full or partial remission.
  
  
  
  In our case series, none of the patients had the full presentation of SAPHO at the first consultation. Some presented with symptoms suggestive for psoriatic arthritis. This explains why diagnosis of SAPHO can be challenging. Full remission was induced in the majority of individuals. Bisphosphonates seem to be a noteworthy treatment option. We suggest a prospective placebo-controlled clinical trial with bisphosphonates to confirm this observation.
authors.affiliation: Department of Rheumatology, University Hospitals Leuven, Herestraat 49, B-3000 Leuven, Belgium.
authors.forename: Tom
authors.initials: T
authors.lastname: Zwaenepoel
authors.affiliation: Department of Rheumatology, University Hospitals Leuven, Herestraat 49, B-3000 Leuven, Belgium. Electronic address: kurt.devlam@uzleuven.be.
authors.forename: Kurt de
authors.initials: K
authors.lastname: Vlam
mesh_terms: D020083:Acquired Hyperostosis Syndrome / Q000175:diagnosis / Q000188:drug therapy*; D000293:Adolescent; D000328:Adult; D050071:Bone Density Conservation Agents / Q000627:therapeutic use*; D002648:Child; D004164:Diphosphonates / Q000627:therapeutic use*; D005260:Female; D006801:Humans; D008297:Male; D008875:Middle Aged; D012189:Retrospective Studies; D012737:Sex Factors; D016896:Treatment Outcome; D055815:Young Adult
chemical_list: D050071:Bone Density Conservation Agents; D004164:Diphosphonates
keywords: Acne; Bisphosphonates; Hyperostosis; Osteitis; Pustulosis; Radiologic imaging; SAPHO; Syndrome of synovitis