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Introduction: Immune checkpoint inhibitors are a class of monoclonal antibodies that are used as a mainstay of immunotherapy for multiple solid organ malignancies. With the recent increase in popularity of these agents, immune-related adverse events including optic neuropathy are becoming more frequently reported. This review aims to explore the association between immune checkpoint inhibitors and optic neuropathy through analysis of incidence, clinical features, investigations, treatment, and patient outcomes., Method: A systematic search of the databases PubMed/MEDLINE, Embase, and CENTRAL was performed from inception to September 2022. Data collection and risk of bias analysis was subsequently conducted in accordance with the PRISMA guidelines., Results: Eleven articles fulfilled the inclusion criteria. The results showed an increased incidence of optic neuropathy among patients receiving immune checkpoint inhibitor therapy compared to the general population. Presentation with painless reduced visual acuity and optic disc swelling was most common. Investigation findings were poorly documented. The only two patients who achieved full resolution of symptoms were treated with oral prednisolone., Conclusion: There is a strong association between immune checkpoint inhibitor therapy and development of optic neuropathy. Although it remains uncommon, the incidence of optic neuropathy in this population exceeds that of the general population. Future research is needed to further characterise the risk profiles of patients who are most likely to develop ICI-associated optic neuropathy, and treatment pathways for these patients.