title: Tocilizumab in refractory adult Still's disease.
abstract: OBJECTIVE
  There is an unmet need for the treatment of adult Still's disease (ASD), the pathogenesis of which may involve interleukin-6 (IL-6). We report the first series of patients with ASD treated with tocilizumab (TCZ), a humanized anti-IL-6 receptor antibody.
  
  
  METHODS
  All ASD patients treated with TCZ in France between July 2006 and July 2009 after failure to all available therapies were included in this cohort study. The main outcome measures were the European League Against Rheumatism (EULAR) improvement criteria and resolution of systemic symptoms at the 3- and 6-month followup periods.
  
  
  RESULTS
  Fourteen patients with refractory ASD were included. At the start of TCZ treatment, despite a mean prednisone dosage of 23.3 mg/day, based on a 28-joint count, mean tender joints were 10.5, mean swollen joints were 7.9, and the mean Disease Activity Score in 28 joints was 5.61. Recurrent systemic involvement, including fever and rash, was present in 7 patients. TCZ was administered at 5-8 mg/kg every 2 or 4 weeks (8 mg/kg/month, n = 9). Eleven patients successfully completed the 6-month study; 1 withdrew due to necrotizing angiodermatitis, another due to chest pain at each TCZ infusion, and a third due to systemic flare. A good EULAR response was observed in 64% of patients (9 of 14) at 3 months and EULAR remission was observed in 57% (8 of 14) at 6 months. Systemic symptoms were resolved in 86% of patients (6 of 7). Moreover, corticosteroid dose was reduced by 56%. No other severe adverse effects occurred.
  
  
  CONCLUSIONS
  TCZ is a promising new treatment for ASD.
authors.affiliation: Le Mans General Hospital, Le Mans, France. xpuechal@ch-lemans.fr
authors.forename: Xavier
authors.initials: X
authors.lastname: Puéchal
authors.forename: Michel
authors.initials: M
authors.lastname: DeBandt
authors.forename: Jean-Marie
authors.initials: JM
authors.lastname: Berthelot
authors.forename: Maxime
authors.initials: M
authors.lastname: Breban
authors.forename: Jean-Jacques
authors.initials: JJ
authors.lastname: Dubost
authors.forename: Olivier
authors.initials: O
authors.lastname: Fain
authors.forename: Jean-Emmanuel
authors.initials: JE
authors.lastname: Kahn
authors.forename: Laurence
authors.initials: L
authors.lastname: Lequen
authors.forename: Maïté
authors.initials: M
authors.lastname: Longy-Boursier
authors.forename: Aleth
authors.initials: A
authors.lastname: Perdriger
authors.forename: Thierry
authors.initials: T
authors.lastname: Schaeverbeke
authors.forename: Eric
authors.initials: E
authors.lastname: Toussirot
authors.forename: Jean
authors.initials: J
authors.lastname: Sibilia
mesh_terms: D000328:Adult; D000368:Aged; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D061067:Antibodies, Monoclonal, Humanized; D015331:Cohort Studies; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D008297:Male; D008875:Middle Aged; D011446:Prospective Studies; D019947:Receptors, Interleukin-6 / Q000037:antagonists & inhibitors / Q000276:immunology; D016706:Still's Disease, Adult-Onset / Q000188:drug therapy* / Q000473:pathology; D055815:Young Adult
chemical_list: D000911:Antibodies, Monoclonal; D061067:Antibodies, Monoclonal, Humanized; D019947:Receptors, Interleukin-6; C502936:tocilizumab