Purpose To report the case of a patient with ocular West Nile virus infection (WNVI) and to describe the demographics, eye characteristics, and treatment of patients with WNVI reported in the literature. Methods Systematic literature search using the PubMed MEDLINE database searching for all cases of ocular WNVI published from inception until October 14, 2023. Inclusion criteria were patients with serologic and/or cerebrospinal fluid diagnosis of WNVI with ocular involvement. Results A total of 60 patients (111 eyes), including the present case, were included. Most patients were males (57%), diagnosed in the United States (77%), and with a mean age at presentation of 54 years. The median time elapsed between the viral prodrome, and eye symptoms was 7 days. Neurologic involvement was present in 47 (78%) patients. Diabetes mellitus was the most frequent systemic comorbidity (45%). Posterior segment findings were present in 107 (96%) eyes. Multifocal chorioretinal lesions (86%), vitreous inflammation (51%), intraretinal hemorrhages (43%), and retinal vasculitis (21%) were the most frequent findings. Fluorescein angiography was performed in 88 (79%) eyes. Fifty-seven (51%) eyes did not receive treatment. Topical and systemic steroids were prescribed to 35% and 28% of eyes, respectively. Conclusion WNVI should be considered as a potential diagnosis in older patients who exhibit posterior uveitis, especially if they have recently experienced flu-like symptoms and have been exposed to mosquitoes. A comprehensive ocular assessment, which includes a dilated fundus examination and ocular imaging studies, can help raise suspicion for this condition even before serological confirmation is obtained.