BACKGROUND : Poor implant positioning has been identified as a factor in early failure of unicompartmental knee arthroplasty. The aim of this study was to report the accuracy of component positioning, and midterm clinical, functional and radiological outcomes following patient-specific instrumented (PSI) unicompartmental knee arthroplasty (UKA). METHODS : A total of 115 PSI-UKA were included. The primary outcomes were UKA survival, complication, and failure rates. Tibial implant positioning was determined using plain radiographs. Functional assessment included Oxford Knee Score (OKS), Forgotten Joint Score (FJS), patient satisfaction, and range of motion (ROM). RESULTS : The survival rate of PSI-UKA was 92% after a mean follow-up of 55 months. The complication and failure rate was 13% and eight percent, respectively. The tibial component was accurately implanted in the desired frontal and sagittal alignment with a minor deviation of 0.3° (SD 1.9°) and 0.4° (SD 2.6°) to the preoperative planning. OKS increased from 24 (SD eight) points to 44 (SD six). FJS was 87 (SD 23) and 89.6% of all patients reported to be satisfied at the final follow-up. Patient satisfaction was negatively correlated with patients' age (p < 0.05). CONCLUSIONS : Excellent accuracy regarding component placement in UKA can be achieved with PSI. However, despite excellent survivorship and clinical outcomes, these data indicate that the PSI system is not superior to conventional UKA implantation methods.