Whereas medical approach to coeliac disease is well defined, treatment of patients who fail to respond to a gluten-free diet remains still problematic. We describe the case of a 68 years DQ-2 positive male who lost response to a strict gluten-free diet after an initial response over a 3-year period. His conditions became critical despite high dose prednisone treatment. After a careful differential diagnosis, the patient was classified as having a type I refractory coeliac disease and a single infusion of infliximab at 5mg/kg was given with excellent clinical results. However, clinical response was lost despite background therapy with azathioprine. Six months after the single infusion an induction therapy with infliximab and, thereafter, maintenance every 8 weeks was administered with excellent clinical results. Since small bowel histology recovered very slowly treatment was continued over the following 2 years with a return to near normal architecture. This case shows that anti-tumour necrosis factor treatment may be used in carefully selected patients with type I refractory coeliac disease.