In reviewing the literature, no studies were found reporting the use of retro running on flat and hilly terrain, which elicited enthesopathy (stress reaction) at the ischial tuberosity. Therefore, this case study of an atypical enthesopathy condition warrants careful scrutiny in order to generate future research. This case study describes the clinical management of a female runner with bilateral patellofemoral pain who self-initiated a program of backward running and stationary bicycling after reading an article about retro running in a runners' magazine. She subsequently developed ischial tuberosity enthesopathy verified by scintigraphy (bone scan). Her symptoms gradually resolved with physical therapy intervention. Eventually, she was able to forward jog 2 miles on flat surfaces without complaint of pain but did not resume retro running. This case not only suggests the need for further research in retro running kinetics and kinematics but highlights the proactive role health professionals must assume in injury prevention.