Purpose: to evaluate the efficacy of monoclonal anti-tumor necrosis factor alpha antibody infliximab in treatment of refractory Wegene’s granulomatosis. Clinical manifestations of Wegener’s granulomatosis at the time of infliximab initiation were: proptosis of both eyes due to progressive retrobulbar granulomas, visual impairment, optic disc edema, progressive diplopia, sinus and renal involvement. The disease was refractory to a treatment with various immunosuppressants, including intravenous cyclophosphamide or high doses of mycophenolate mofetil in combination with corticosteroids. Methods: observational case report. Results: the patient received six infusions of infliximab (3mg/kg) in addition to low doses of oral cyclophosphamide and corticosteroids. The interval between the first two infusions was 1 week. The interval between the second and third infusions was 4 weeks, thereafter 8, 12 and 34 weeks. Following the third infusion of infliximab the remission of Wegener’s granulomatosis was seen. After the regression of retrobulbar granulomas visual acuity increased, ocular motility improved, diplopia and optic disc edema decreased. Remission has been maintained for 17 months. Infliximab was well tolerated during the shortterm follow-up. Conclusion: Infliximab successfully induced remission in case of sightthreatening Wegener’s granulomatosis in our patient. It seems that infliximab provides more effective therapeutic option in treatment of Wegener’s granulomatosis refractory to standard immunosuppression and offers better perspectives for patients with previously poor prognosis. Furthermore, it enables reducing the exposure to immunosuppressive drugs.

        Key words: infliximab, TNF-α, Wegener’s granulomatosis