Background: Progressive supranuclear palsy (PSP) is a rare neurodegenerative disease and currently no effective symptomatic or neuroprotective treatment is available for PSP. Deep brain stimulation (DBS), as a neurosurgical procedure, plays an important role in a range of neurological and psychiatric disorders. However, there are no systematic investigations about the DBS in PSP patients. Objective: We performed a systematic review and meta-analysis to evaluate the safety and efficacy of DBS for PSP. Methods: PubMed, EMBASE, Cochrane library, Chinese National Knowledge Infrastructure and Wan Fang databases were systematically searched without time restrictions. We assessed the data between DBS-OFF and DBS-ON groups, as measured by the Unifed Parkinson Disease Rating Scale (UPDRS). Results: Of 154 identified studies, 13 were eligible and were included in our meta-analysis (N = 36 participants). A reduction of UPDRS Ⅲ scores under DBS-ON conditions was observed, but the differences yielded no statistical significance. Conclusion: Since part of PSP patients could benefit from DBS, we speculate that DBS may become a safe and promising tool for PSP in axial symptoms as well as non-motor symptoms though further investgations are needed. Our findings will provide design strategies for following clinical trials and ultimately help improve the clinical application of DBS in PSP patients.