A 25-year-old man with a family history of essential hypertension was detected to have hypertension for two years. To rule out secondary cause, he underwent the usual tests and was found to be normal. After initial control for two years, he was recently requiring two antihypertensives even with excellent compliance and strict lifestyle measures. One day he presented to our emergency room with an acute stroke involving the left corona radiata, frontal, and temporal regions, and an angiogram revealed moyamoya disease. We noted that he never had any neurological symptoms earlier and hence never underwent brain imaging before this incident.