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Moyamoya disease (MMD) is a rare degenerative stenosis and occlusive cerebrovascular illness. It is characterized by cerebral ischemia and/or cerebral hemorrhage as the two main clinical signs. It is a common cause of stroke in both children and adults. Several recent studies illustrated the crucial role of microRNAs (miRNAs) in the pathophysiology of MMD via the regulation of endothelial cells and smooth muscle cells. Furthermore, other studies highlighted the decisive role of miRNAs in the underlying molecular pathophysiological mechanisms in MMD via regulation of cellular proliferation, angiogenesis, extracellular matrix remodeling, and vascular inflammation. This review aims to explore the involvement of miRNAs in MMD pathogenesis and to assess their potential use as biomarkers for stroke risk and their utility as therapeutic targets for the treatment of MMD.