Moyamoya disease (MMD) is a rare cerebral vasculopathy with limited information on the associated cognitive and emotional sequelae, particularly presenting in a psychiatric setting. We present the case of a 25-year-old female with a history of MMD and depressive disorder. She underwent revascularization surgery following an ischemic stroke at age 15 years. Magnetic resonance imaging at the time of the current assessment revealed markedly abnormal appearance in the anterior cerebral circulation with extensive periventricular collateral vasculature involvement. Neuropsychological assessment data revealed deficits in inhibition and problem-solving, consistent with frontal lobe dysfunction. We discuss the importance of further research on the neuropsychological sequelae of MMD.