Duchenne muscular dystrophy (DMD) is a progressive form of muscular dystrophy caused by mutations in the dystrophin gene. Patients with DMD are more likely to have cerebral infarction than normal populations, possibly due to low ejection fraction and cardiomyopathy, and also higher epilepsy prevalence. Careful history taking and neurological examination are needed for differentiating new symptoms from preexisting weakness in DMD. Here, we present a young male with DMD and acute ischemic stroke followed by recurrent seizures.