Background The efficacy of anti-TNF treatment for patients with severe forms of Behcet's disease (BD) is well established (ref), but long term data on the outcome after cessation of such treatment are lacking.
Objectives To examine whether sustained long term remission of severe BD is feasible after cessation of successful anti-TNF treatment.
Methods This retrospective longitudinal outcome study was conducted in December 2016 and included all patients with severe BD refractory to conventional immunosuppressive therapy who were considered complete responders to continuous anti-TNF treatment in our center, the first being treated in 2000. Study's endpoint was the proportion of patients achieving sustained complete remission of BD for at least 3 years after cessation of the anti-TNF agent.
Results A total of 28 patients in whom infliximab and/or adalimumab treatment was given, always combined with azathioprine unless not tolerated (n=2), and discontinued anytime before December 2013 were eligible for analysis. Following cessation of successful anti-TNF treatment (median duration of 2 years) 13/28 patients achieved the study's end-point. The main reason for anti-TNF administration was sight-threatening ocular disease (n=12) or intestinal disease (n=1). The remaining 15 patients relapsed within 1.5 year (main reason for anti-TNF: ocular disease, n=9; neuro-BD, n=2; severe mucocutaneous disease, n=3; intestinal disease, n=1; median treatment duration of 24 months); 12/15 were successfully re-treated with anti-TNF. So far, 3 of them (ocular disease, n=2; neuro-BD, n=1) have achieved the study's end-point (median re-treatment duration of 2 years). Overall, our 16 patients who achieved the study's end-point (57%) are in complete disease remission ranging from 3 to 12 years (5.7 years, median). Nine patients with severe ocular disease are currently any drug-free (32%), whereas the 7 remaining patients are on low doses of conventional immunosuppressive therapy (25%). Notably, those patients on drug-free remission had shorter median disease duration at initiation of anti-TNF treatment, compared to the remaining patients (1 versus 3 years, respectively).
Conclusions Sustained drug-free remission for many years after cessation of successful anti-TNF treatment is feasible in some patients with severe BD. Since anti-TNF-induced “cure” can never be differentiated from a spontaneous remission by natural history, further studies should examine whether early anti-TNF treatment must be intended for every patient with severe BD.
Hatemi G, et al. EULAR recommendations for the management of Behçet disease. Ann Rheum Dis. 2008 Dec;67(12):1656–62.
Disclosure of Interest None declared