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Introduction: SMART syndrome is a rare complication of brain radiotherapy. This is the first described presentation of SMART syndrome with metamorphopsia, which responded to aspirin, verapamil, and high-dose L-arginine therapy. Case Presentation: A 43-year-old man presented with 3 weeks of migraine headaches with metamorphopsia and complex visual hallucinations affecting the left lower quadrant of both visual fields. This occurred on a background of high-dose radiotherapy for right cerebellar astrocytoma 32 years ago. MRI brain demonstrated unilateral gyriform enhancement and FLAIR hyperintense cortical swelling in the right occipital lobe consistent with SMART syndrome. Conclusion: Unusual presentations of SMART syndrome exist and require consideration in all patients with focal neurological deficit post-brain radiotherapy. Validated diagnostic and treatment modalities for SMART syndrome are urgently required.