Orofacial mesenchymal stromal cells (OFMSCs) are mesenchymal stromal cells isolated from the oral and facial regions, which possess typical mesenchymal stromal cell features such as self-renewing, multilineage differentiation, and immunoregulatory properties. Recently, increasing studies have been carried out on the neurotrophic and neuroregenerative properties of OFMSCs as well as their potential to treat neurological diseases. In this review, we summarize the current evidence and discuss the prospects regarding the therapeutic potential of OFMSCs as a new approach to treat different neurological diseases and injuries.