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This single-case study design describes the impact of training on a commercially available motor-assisted elliptical trainer on walking endurance and oxygen cost of self-selected comfortable (SSC) gait speed for an individual with progressive supranuclear palsy (PSP).A 67-year-old man with PSP and difficulties with ambulation, movement initiation, falls, and balance participated in a structured intervention on a motor-assisted elliptical trainer with body weight support (30 minutes, 3 d/wk, total of 24 sessions). The participant was systematically progressed across the training sessions by manipulating the motor-assisted elliptical training parameters (e.g., speed, step length, motor assistance, and body weight support). Walking endurance and oxygen cost of SSC gait speed were measured pretraining, posttraining, and at 1-month follow-up.The participant's 6-minute walk test (6MWT) distance and oxygen cost improved after training. One month of detraining revealed sharp declines in the 6MWT distance and worsening of his Freezing of Gait questionnaire score. However, improved oxygen cost was sustained at 1-month follow-up. This case study is the first to document outcomes from a motor-assisted elliptical training program for an individual with PSP. Improved walking distance and oxygen cost suggest possible benefits of structured exercise for an individual with PSP.