Zoledronic acid is recommended for patients with osteoporosis. To report a case of unilateral anterior uveitis after zoledronate infusion. An osteoporotic patient presented with pain, visual loss, hyperemia, photophobia, and watering from the left eye after zoledronate infusion. Circumcorneal injection, keratic precipitates, cells, and flare suggested anterior uveitis. Her symptoms resolved completely after 20 days of prednisolone acetate with atropine eye drops. Uveitis is a rare complication of zoledronic acid with an unclear mechanism. Proinflammatory cytokines may play a role in pathogenesis. Zoledronic acid may be associated with rare but serious inflammatory ocular adverse drug reactions.