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Although the short-term effects of early, aggressive DMARD therapy for patients with juvenile idiopathic arthritis (JIA) are positive, the long-term effects have not been assessed.To assess patients who were involved in a previous study of early, aggressive sulfasalazine treatment for JIA to determine if there are long-term benefits associated with this therapeutic strategy.This study was a follow-up analysis of 61 patients with JIA who had participated in a 24-week, multicenter, double-blind, randomized, placebo-controlled trial of sulfasalazine ∼9 years earlier. Patients and their medical records were examined once by a blinded investigator.Outcome measures included a physician's global assessment of disease activity on a 100 mm visual analog scale, a graded physician's global assessment of disease activity score, discomfort as assessed by the 100 mm visual analog scale for pain and overall well-being, and erythrocyte sedimentation rate. Overall outcome was assessed with an adaptation of the American College of Rheumatology (ACR) Pediatric 30 definition of improvement, which utilized data on number of joints with active disease, number of limited joints, physician's global assessment of disease activity score and erythrocyte sedimentation rate; patients were classified as improved if they had >30% improvement in at least 3 of these 4 variables. Patients were classified as noncompliant if they reported that they had not taken their prescribed dose of DMARDs for at least 6 months on any occasion between the initial randomized controlled trial (RCT) and the follow-up study.Over 90% of the participants in the original RCT were included in the follow-up study; 29 patients in the sulfasalazine cohort (n = 32) and 27 patients in the placebo group (n = 29) were treated with DMARDs immediately after the original RCT trial. At the time of their follow-up appointments, 17 patients from the sulfasalazine cohort and 21 patients from the placebo cohort were receiving DMARDs. The median duration of sulfasalazine treatment was significantly longer (5.2 years versus 2.5 years, P = 0.02) for patients who had received placebo in the original RCT compared with those who had received sulfasalazine. Patients in the original sulfasalazine cohort had a decreased number of joints with active disease (P<00.01), improved overall well-being (P <0.01) and longer episodes of clinical remission off medication (P <0.01) compared with patients who had originally received placebo. Additionally, compared with the placebo cohort, a greater number of patients in the sulfasalazine group improved according to the ACR Pediatric 30 definition (P <0.01), had episodes of clinical remission off medication (P <0.01) and were experiencing clinical remission off medication at the time of their follow-up appointment (P <0.01). Noncompliant patients (41%) had significantly fewer episodes of clinical remission off medication (P = 0.007) and more operations (P = 0.03) compared with compliant patients.Delayed initiation of sulfasalazine treatment for JIA had long-term negative effects, and noncompliance with DMARD therapy was very common.