Progressive supranuclear palsy (PSP) is characterized by recurrent falls caused by postural instability, and a backward gait is considered beneficial for postural instability. Furthermore, a recent approach for rehabilitation combined with gait-oriented synchronized stimulation using non-invasive transcranial patterned stimulation could be promising for balance function. Here, we present a case of PSP with backward gait training combined with gait-synchronized transcranial alternating current stimulation (tACS). A 70-year-old woman with PSP-Richardson’s syndrome underwent backward gait training combined with synchronized cerebellar tACS. Initially, she underwent short-term intervention with combined training of backward gait with synchronized cerebellar tACS, asynchronized, or sham stimulation according to the N-of-1 study design. Synchronized tACS training demonstrated a decrease in postural instability, whereas asynchronized or sham stimulation did not. The additional long-term interventions of combined backward gait training with synchronized cerebellar tACS demonstrated further decrease in postural instability with improvements in gait speed, balance function, and fall-related self-efficacy in daily life. The present case describes a novel approach for motor symptoms in a patient with PSP. Backward gait training with synchronized cerebellar tACS may be a promising therapeutic approach.