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Diagnostic dilemma of ocular lymphoma. We report a very unusual presentation of primary intraocular lymphoma masquerading as anterior uveitis with atypical symptoms. A 68-year-old man, initially presented with a 4-day history of painless, left blurred vision. Examination revealed 2 or more anterior chamber cells, mutton-fat keratic precipitates and posterior synechiae. Treatment was initiated, the eye settled, and the patient was discharged. He re-presented 7 months later with hypopyon, a hazy cornea, fibrin deposits and a narrowed anterior chamber angle. Over the coming weeks, it became apparent that the temporal iris was thickened, involving the angle, and his intraocular pressure increased despite topical dorzolamide. Following advice from a tertiary centre and referral to a regional ocular oncology centre, a transscleral biopsy was performed and suggested a ciliary body melanoma. Enucleation was advised and performed, with histological examination revealing features consistent with an ocular diffuse large B-cell lymphoma not involving the optic nerve, sclera or cornea. This is the first ever published report of a case of ocular lymphoma masquerading in this way.