Background: This is a rare case of piriformis syndrome. We discuss the patient’s symptoms, imaging study results, laboratory examination findings, and prescribed treatment in an attempt to determine the possible mechanisms or reasons for this patient’s development of piriformis syndrome. Methods: The patient is a 22-year-old female soldier who was admitted for lower back pain with soreness radiating to her right buttock and right lower extremity. We found that she had leg length discrepancy under X-ray finding. She was diagnosed piriformis syndrome by physical examination and Magenetic Resonance Image. Results: We performed scanography for lower extremities revealed a left femur length of 42.7 cm, a right femur length of 43.3 cm, a left tibia length of 33.2 cm, and a right tibia length of 33.6 cm;her left lower extremity was 1 cm shorter than her right. The MRI of sacrum also reveals hypertrophy of right piriformis muscle. Conclusions: Piriformis syndrome might be caused by short-term compensation in congenital leg length discrepancy. The patient with leg length discrepancy changed posture when standing, walking, and running. These compensation postures could lead to abnormality position of bones where there is insertion of the muscle.