Background: To report on anti-CD20 antibody therapy in a patient with uveitis refractive to immunosuppression therapy. Methods: Case report with ophthalmoscopic, optical coherence tomography and fluorescein-angiographic findings. Results: A 49-year-old woman was suffering from bilateral, noninfectious chronic anterior uveitis refractive to corticosteroids and immunosuppressive drugs. Bilateral visual acuity was 20/100 due to cataract and cystoid macular edema (CME). After treatment with rituximab, vision and CME improved, and uveitis was stable until the final visit (follow-up at 12 months). Conclusion: The case report suggests that rituximab may be helpful for selected patients with chronic anterior uveitis refractive to corticosteroids and immunosuppressive medication.

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