A 35-year-old patient received omalizumab (300 mg twice a month) for 2 years for a severe atopic keratoconjunctivitis (AKC) in order to reduce the risk for steroid-induced keratitis. After 4 months, quality of life and ocular symptoms improved, and the use of oral steroids was strongly reduced. The treatment was well tolerated. This observation suggests that omalizumab may be a good option for the treatment of severe AKC, especially to avoid side effects of immunosuppressive treatments, as suggested for other allergic diseases. Specific trials should be designed for allergic eye diseases.