Behcet's disease is a chronic relapsing multisystem vasculitis with 49% of cases involving the CNS. Recently there have been two reports of neuro-Behcet's disease (NB) successfully treated with the tumour necrosis factor alpha (TNF alpha) monoclonal antibody infliximab. We describe a patient with longstanding NB who was poorly responsive to azathioprine, cyclosporin, thalidomide and methotrexate. She showed a remarkable response when treated with the recombinant human TNF alpha receptor protein, etanercept. To the best of our knowledge this is the first report of NB successfully treated with etanercept. (c) 2006 Elsevier B.V. All rights reserved.