title: Long-term outcome of anti-tumor necrosis factor alpha blockade in the treatment of juvenile spondyloarthritis.
abstract: OBJECTIVE
  A significant proportion of patients with juvenile spondyloarthritis (JSpA) are refractory to treatment with established medications. The objective of this study was to assess long-term efficacy of treatment with anti-TNF agents in patients with JSpA.
  
  
  METHODS
  An observational study of 16 patients with JSpA from 3 centres treated with infliximab (n=10) and etanercept (n=6) was performed, with a median follow-up period of 7.2 years. Prospective data was collected according to a standardized protocol. Outcomes examined were TEC, TAJC, markers of inflammation (ESR, CRP), functional assessments (C-HAQ, BASDAI, BASFI), and ongoing requirement for anti-TNF treatment.
  
  
  RESULTS
  13/16 patients (83%) had achieved clinical remission 6 months into the treatment. Improvement was sustained over time, with a median TAJC and TEC of 0 at any time point after 6 weeks. 6/16 patients (38%) showed a flare of arthritis after a median of 3.5 years. Two patients with hip disease prior to treatment required an arthroplasty 3 and 8 years post anti-TNF initiation. Patients showed progression of sacroiliitis with median modified New York score of 1 (range 0-3) at time of diagnosis and 3 (range 0-4) at last follow-up (p=0.002). Median BASDAI at last follow up was 1.6, median BASFI 3.1. Two patients developed transient reactions (one generalised, one local); no patient developed other adverse effects during the study.
  
  
  CONCLUSIONS
  Anti-TNF treatment in JSpA refractory to standard treatment results in good long-term disease control except for pre-existing hip disease. However, radiographic evidence suggests inferior efficacy for control of sacroiliac joint disease.
authors.affiliation: Division of Rheumatology, The Hospital for Sick Children, University of Toronto, Canada, and German Center for Paediatric and Adolescent Rheumatology, Garmisch-Partenkirchen, Germany. huegle.boris@rheuma-kinderklinik.de.
authors.forename: B
authors.initials: B
authors.lastname: Hugle
authors.forename: R
authors.initials: R
authors.lastname: Burgos-Vargas
authors.forename: R D
authors.initials: RD
authors.lastname: Inman
authors.forename: F
authors.initials: F
authors.lastname: O'Shea
authors.forename: R M
authors.initials: RM
authors.lastname: Laxer
authors.forename: J
authors.initials: J
authors.lastname: Stimec
authors.forename: K
authors.initials: K
authors.lastname: Whitney-Mahoney
authors.forename: M
authors.initials: M
authors.lastname: Duvnjak
authors.forename: M
authors.initials: M
authors.lastname: Anderson
authors.forename: S M L
authors.initials: SM
authors.lastname: Tse
mesh_terms: D000293:Adolescent; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D018501:Antirheumatic Agents / Q000627:therapeutic use*; D001171:Arthritis, Juvenile / Q000188:drug therapy; D002648:Child; D000068800:Etanercept; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D007074:Immunoglobulin G / Q000627:therapeutic use*; D000069285:Infliximab; D008137:Longitudinal Studies; D008297:Male; D018124:Receptors, Tumor Necrosis Factor / Q000627:therapeutic use*; D058566:Sacroiliitis / Q000188:drug therapy; D025241:Spondylarthritis / Q000188:drug therapy*; D016896:Treatment Outcome; D014409:Tumor Necrosis Factor-alpha / Q000037:antagonists & inhibitors*
chemical_list: D000911:Antibodies, Monoclonal; D018501:Antirheumatic Agents; D007074:Immunoglobulin G; D018124:Receptors, Tumor Necrosis Factor; D014409:Tumor Necrosis Factor-alpha; D000069285:Infliximab; D000068800:Etanercept