Background Patient-specific instrument (PSI) may theoretically make total ankle arthroplasty (TAA) more accurate. Several studies have reported the outcomes of PSI TAA. The aim of this study is to systematically review the literature of PSI TAA. Methods PubMed, Embase, Web of Science, and Cochrane Library databases were systematically reviewed according to the Preferred Reporting Items for Systematic Reviews and Meta-analysis (PRISMA) guidelines for PSI TAA. The quality of the included studies was evaluated according to Methodological Index for Non-Randomized Studies (MINORS). Result Nine articles were ultimately included in the systematic review. The implant position and function outcome of TAA was similar between PSI and SI. Prediction accuracy of implant size remained great difference. PSI can shorten the operative time and fluoroscopy time. The quality of current studies on PSI TAA is insufficient to produce high-level evidence. Conclusion PSI can get similar implant position and clinical outcome in TAA compared to SI, but current evidence is not strong enough to evaluate PSI TAA.