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We report a case of a 4-year-old boy with a history of global developmental delay presenting with recurrent sudden onset of weakness and spontaneous recovery and refractory focal seizures. The findings of his brain magnetic resonance imaging revealed that the underlying cause of his unexplained neurological deficits was moyamoya disease (MMD). Revascularization can cure MMD-associated transient ischemic attacks and seizures. Proper control of seizures and blood pressure and early use of antiplatelet treatment before the surgery may lead to better outcomes.