An 11-year-old female felt discomfort in her head, and left hemispheric syndrome occurred shortly thereafter. At presentation, her National Institutes of Health stroke scale (NIHSS) score was 13, and a magnetic resonance imaging scan revealed acute brain infarction in the left thalamus. She was immediately treated with the intravenous administration of tissue plasminogen activator (IV t-PA) followed by edaravone, a free radical scavenger. Two hours after IV t-PA, her symptoms dramatically resolved and her NIHSS score decreased to 5. No adverse events were observed. She was the youngest patient treated with IV t-PA in Japan, and would be the youngest treated in most developed countries. An optimal treatment for stroke in children has not been established, and this case highlights the urgent need to examine the safety and efficacy of IV t-PA and edaravone therapy for ischemic stroke in children.