Prader-Willi syndrome (PWS) is a genetic disorder characterized by specific physical and behavioral abnormalities and considered the most commonly known genetic cause of morbid obesity in children. Recent studies indicate that patients suffering from this syndrome have significant problems in skill acquisition, muscle force, cardiovascular fitness, and activity level. In this study, we report an obese adolescent PWS patient of poor aerobic fitness compared with 13 obesity adolescents, and great improvement in cardiopulmonary exercise test (CPET) outcomes of the PWS patient measured after two weeks of physical exercise training programs.