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Intravesical Bacillus Calmette-Guérin (BCG) instillation has become one of the mainstays of adjunctive therapy in the treatment of superficial bladder cancer. Ophthalmologic complications are rare, but few cases are reported in the literature.Retrospective observational case report.The authors report a case of unilateral Mycobacterium bovis BCG endophthalmitis after intravesical BCG instillations. Despite appropriate systemic antituberculous and corticosteroid therapy, the patient almost completely lost sight in the affected eye. This is the fourth case in the literature of proven M. bovis endophthalmitis suggesting a direct choroidal mycobacterial infection and not only a hypersensitivity immunologic reaction as previously suggested.This case highlights the direct choroidal mycobacterial infection of the disease after BCG instillations for bladder cancer and failure of treatment despite culture-proven drug sensitivity, thus suggesting the need to revaluate adequate treatment to avoid loss of vision.