This is a retrospective study about the long-term efficacy of rituximab in patients with severe juvenile idiopathic arthritis (JIA)-associated uveitis. A total of eight patients (15 eyes) were recruited in this study in a single tertiary referral centre from Italy. Rituximab was given at the dosage of 1000mgs per infusion on days one and 15 and then at three to six months. The clinical responses to the treatment including a decrease in uveitis activity, visual acuity changes, reduction of local and systemic steroid and / or immunosuppression, and occurrence of adverse events were assessed in this article. The mean age of treated patients was 22.8 ±5.5 years. The mean ocular disease duration was 17.7 years; the mean ± standard deviation follow-up time on rituximab was 44.75±4.9 months. The mean number of rituximab infusions received was 8.7 (range 6-12). Only two patients on rituximab did not respond to the treatment and were discontinued due to inefficacy on the arthritis. The decrease in uveitis activity was evident four to five months after the first infusion. Systemic steroid and immunosuppression used in association with rituximab were discontinued in five patients at the end of the follow-up period. None of the patients experienced visual worsening during the follow-up. No drug-related complications were encountered in this study. The two patients with arthritis who did not respond to rituximab were switched to golimumab. The authors conclude that rituximab may be a new treatment option with a convenient dosing schedule for the treatment of patients with autoimmune diseases, particularly those who have not previously responded to TNF-α blockers. In addition, they advise the results have to be interpreted with caution in the context of the retrospective design and nature of the study, and also the small numbers and heterogeneity of the patients and lack of the controlled group in this study. They propose further randomised, prospective trials of rituximab in a larger group of patients are needed to evaluate the better efficacy dosing regime and safety of this treatment in patients with JIA-associated uveitis resistant to previous conventional treatments.