Cytomegalovirus retinitis (CMVR) is a rare complication of systemic immunosuppressive therapy in patients with rheumatic disorders. We describe a 44-year-old man with Wegener’s granulomatosis who was treated with cyclophosphamide and methylprednisolone and who subsequently developed bilateral CMVR. In spite of discontinuation of immunosuppressive therapy and administration of systemic ganciclovir, retinitis recurred and maintenance therapy was needed. In conclusion, increased awareness of this potentially serious complication of immunosuppressive therapy is critical. If immune recovery is slow after discontinuation of immunosuppressive agents, prolonged antiviral therapy is required in order to prevent recurrences of CMVR.