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Background: Axitinib is a selective inhibitor of vascular endothelial growth factor receptors and is used in the treatment of many malignancies. Herein, we reported a rare case with axitinib-induced retinal changesand associated toxicity., Case Presentation: A forty-five-year-old female presented with blurred vision who had been taking 7 mg of Axitinib bid for 5 months. Initial Best Corrected Visual Acuity (BCVA) was 20/32 at the right and counting fingers at the left eye. Funduscopic examination revealed bilaterally widespread intraretinal hemorrhages, cotton-wool spots, and hard exudates with a star-like appearance at the macula. The optical coherence tomography revealed central macular edema. There was hyperreflective edema in the inner layers, exudates in the middle retinal layers, and subfoveal subretinal fluid. Fundus fluorescein angiography revealed localized ischaemic findings in the early phase and multifocal perivascular ink-blot fluorescein leakage in the middle and late phases. Axitinib treatment was discontinued immediately, and at the third month of follow-up, the macular edema and fundus findings improved with a final BCVA of 20/20 at the right and 20/32 at the left eye., Conclusion: Considering the ocular side effects of the patients receiving axitinib is crucial to prevent any potentially persistent visual loss., (Copyright© Bentham Science Publishers; For any queries, please email at epub@benthamscience.net.)