A 79-year-old male patient was followed for unilateral uveitis with 3 attacks in 10 months, despite initial improvement with steroid therapy. The patient had visual acuity (VA) of counting fingers in right eye, hypopyon and vitritis with no chorioretinal lesions. The left eye was normal. The patient was evaluated for intraocular foreign body, intraocular lymphoma and associated systemic disease and malignancy. Computed tomography of the abdomen showed a mass in the bladder. Biopsy confirmed bladder carcinoma. After resection of the mass, intraocular inflammation improved completely and no attack was noted in the follow-up. In his last examination, two years after the operation, VA was light perception; seclusio pupilla and mature cataracts were seen on biomicroscopy. There was no sign of vitritis on ocular ultrasonography. Evidence is discussed that suggests a link and potential etiology between refractory uveitis with hypopyon and bladder carcinoma. This is the first case of unilateral recurrent uveitis with hypopyon as the initial presenting sign of bladder carcinoma