title: Rheumatic and musculoskeletal features of Whipple disease: a report of 29 cases.
abstract: OBJECTIVE
  Whipple disease is a rare infection caused by Tropheryma whipplei. Although patients commonly complain of osteoarticular involvement, musculoskeletal manifestations have been poorly described. We report cases of Whipple disease with rheumatic symptoms and describe their clinical presentation, modes of diagnosis, and outcomes.
  
  
  METHODS
  This retrospective multicenter study included patients with Whipple disease diagnosed and referenced between 1977 and 2011 in 10 rheumatology centers in France and Italy.
  
  
  RESULTS
  Twenty-nine patients were included. The median age was 55 years. The median time to diagnosis from first symptoms was 5 years. Polyarthritis was the most frequent presentation (20/29), and was most often chronic, intermittent (19/29), seronegative (22/23), and nonerosive (22/29). In all cases, the symptoms had led to incorrect diagnosis of inflammatory rheumatic disease and immunosuppressants, including biotherapy, were prescribed in most cases (24/29) without success. The diagnosis of Whipple disease was made by histological analysis, molecular biology tests, or both in 21%, 36%, and 43% of the cases, respectively. Duodenal biopsies were performed in most cases (86%). Synovial biopsies were performed in 18% of cases, but all contributed to diagnosis. The clinical outcomes after antibiotic therapy were good for all patients.
  
  
  CONCLUSIONS
  Polyarthritis is the main feature observed in cases of Whipple disease; it is seronegative and associated with general and gastrointestinal symptoms. The molecular analysis of duodenal tissue and/or other tissues remains the method of choice to confirm the diagnosis. Reducing the time to diagnosis is important because severe late systemic and fatal forms of the disease may occur.
authors.affiliation: From the Paris Descartes University, Rheumatology A Department and Rheumatology B Department, Cochin Hospital, AP-HP, Paris; Rheumatology Department, Le Mans Hospital; Rheumatology Department, CHU Angers; Rheumatology Department, CHU Clermont-Ferrand; Rheumatology Department, Bichat Hospital, AP-HP, Paris; Rheumatology Department, CHU Nantes; Unità di Reumatologia, Azienda Ospedaliera Universitaria Integrata, Verona, Italy; Rheumatology Department, CHU Strasbourg; and the Rheumatology Department, Teaching Hospital of Lapeyronie and University of Montpellier, France.
authors.forename: Marine
authors.initials: M
authors.lastname: Meunier
authors.forename: Xavier
authors.initials: X
authors.lastname: Puechal
authors.forename: Emmanuel
authors.initials: E
authors.lastname: Hoppé
authors.forename: Martin
authors.initials: M
authors.lastname: Soubrier
authors.forename: Philippe
authors.initials: P
authors.lastname: Dieudé
authors.forename: Jean Marie
authors.initials: JM
authors.lastname: Berthelot
authors.forename: Paola
authors.initials: P
authors.lastname: Caramaschi
authors.forename: Jacques-Eric
authors.initials: JE
authors.lastname: Gottenberg
authors.forename: Laure
authors.initials: L
authors.lastname: Gossec
authors.forename: Jacques
authors.initials: J
authors.lastname: Morel
authors.forename: Emilie
authors.initials: E
authors.lastname: Maury
authors.forename: Julien
authors.initials: J
authors.lastname: Wipff
authors.forename: André
authors.initials: A
authors.lastname: Kahan
authors.forename: Yannick
authors.initials: Y
authors.lastname: Allanore
mesh_terms: D000328:Adult; D000368:Aged; D000900:Anti-Bacterial Agents / Q000627:therapeutic use; D001168:Arthritis / Q000175:diagnosis* / Q000188:drug therapy / Q000382:microbiology*; D001170:Arthritis, Infectious / Q000175:diagnosis / Q000188:drug therapy / Q000382:microbiology; D042241:Early Diagnosis; D005260:Female; D005602:France; D005767:Gastrointestinal Diseases / Q000175:diagnosis / Q000188:drug therapy / Q000382:microbiology; D006801:Humans; D007166:Immunosuppressive Agents / Q000627:therapeutic use; D007558:Italy; D008297:Male; D008875:Middle Aged; D009140:Musculoskeletal Diseases / Q000175:diagnosis / Q000188:drug therapy / Q000382:microbiology; D012189:Retrospective Studies; D054851:Tropheryma; D008061:Whipple Disease / Q000175:diagnosis* / Q000188:drug therapy / Q000382:microbiology*
chemical_list: D000900:Anti-Bacterial Agents; D007166:Immunosuppressive Agents
keywords: ARTHRITIS; INFECTION; WHIPPLE DISEASE