It remains an unsolved problem in the treatment of patients with refractory focal epilepsy originating from the motor cortex since resection surgery can result in significant morbidity. Neurostimulation has emerged as an effective method for treating patients who are not suitable for conventional surgical procedures due to its relative safety, reversibility, and lower risk of complications. The subthalamic nucleus (STN) has been shown to be a potential target for treating refractory motor seizures. Here, we report a favorable outcome of unilateral deep brain stimulation (DBS) of the STN for a patient with drug-resistant focal myoclonic seizures during a 5-year follow-up period. 2020 Annals of Translational Medicine. All rights reserved.