TNFalpha (tumor necrosis factor alpha) plays a central role in the development of thyroid associated ophthalmopathy (TAO). We describe and document by ophthalmic (CAS and NO SPECS scales) and radiological (MRI) evaluation a positive effect of anti-TNFalpha antibody (infliximab) administration on active TAO in a 58 years old woman with Graves' disease. The single dose of infliximab administration resulted in a dramatic reduction of inflammation studies and improvement of visual function as measured by MRI and CAS and NO SPECS scales, without noticeable short-term side effects. A randomized prospective study is needed to determine whether infliximab proves to be sufficiently effective in reducing the inflammatory symptoms of TAO, and whether it can be administered safely for a prolonged period without side effects.