Purpose: To report the outcomes of infliximab treatment of birdshot retinochoroidopathy (BSRC) refractory to conventional immunomodulatory therapy. Design: Retrospective case series. Participants: Twenty-two refractory birdshot retinochoroidopathy patients (44 eyes) who received infliximab between July 2005 and June 2012 were identified by retrospective chart review. Methods: All patients received 4 to 5 mg/kg infliximab at 4- to 8-week intervals. Data regarding patient demographics, use of immunosuppressive drugs, biologic agents, and reason for conventional therapy discontinuation were gathered. Disease activity markers, including signs of ocular inflammation, fluorescein angiography evidence of retinal vasculitis or papillitis, indocyanine green angiography evidence of active choroiditis, electroretinography parameters indicative of stable or worsening of retinal functions, and optical coherence tomography findings indicative of static or worsening macular edema were recorded. Main Outcome Measures: Abolition of all evidence of active inflammation, visual acuity (VA), presence of cystoid macular edema at 6 months and 1 year, and adverse responses to infliximab. Results: Mean duration of disease before starting infliximab was 58.6 months. Before infliximab therapy, all patients received and failed conventional immunosuppressive therapy. Ten patients had received another biologic agent. After initiating infliximab, control of inflammation was achieved in 81.8% at 6 months and in 88.9% at the 1-year follow-up. Three patients had active inflammation during therapy. The rate of cystoid macular edema decreased from 22.7% at baseline to 13.9% at 6 months and 6.7% at 1 year after receiving the drug. Initial VA of 20/40 or better was found in 34 eyes (84.1%). At 6 months and 1 year, 91.7% and 94.4% of eyes, respectively, had VA of 20/40 or better. Six patients had adverse events; infliximab therapy was discontinued in these patients because of neuropathy, drug-induced lupus, allergic reaction, or fungal infection. Conclusions: The data suggest that infliximab is effective for controlling inflammation in otherwise treatment-refractory cases of BSRC. Financial Disclosure(s): The author(s) have no proprietary or commercial interest in any materials discussed in this article. Ophthalmology 2013;120:588-592 (C) 2013 by the American Academy of Ophthalmology.