Infliximab is a monoclonal antibody that targets TNF-alpha and has been shown to be effective for the management of steroid-dependent or refractory Crohn's disease. It is an effective therapy in adult patients, but experience in children is limited. We report a case of Crohn's disease which was refractory to the conventional treatment. A 14-year-old boy was admitted to the hospital with arthralgia and oral and perianal lesions. On physical examination his body weight was below the 3rd percentile, and height was between the 3rd-10th percentiles. He had elevated erythrocyte sedimentation rate and C-reactive protein and decreased hemoglobin, hematocrit and albumin levels. Barium enema and computerized abdominal tomography revealed a markedly distended small bowel with a narrowed area just above the ileocecal valve and terminal ileum. There was no mucosal pathology in his colonoscopic study. A regimen of prednisolone was begun with a diagnosis of Crohn's disease. In the first month of therapy the patient experienced progressive worsening of his symptoms, and azathioprine was added to the treatment in the second month. As he had exacerbation of his symptoms and worsening laboratory tests, infliximab infusions (5 mg/kg/d) were administered intravenously (at 0, 2 and 6 weeks) at the end of the 8th week. At the 6th week of treatment including two infusions of infliximab at 0 and 2 weeks, clinical and laboratory response occurred. The only side effect of the treatment was pneumonia, which was seen after the 6th week of the therapy. In conclusion, infliximab appears to be an effective and safe therapy for childhood refractory Crohn's disease.