We report the case of a pathological mediopatellar plica found in the right knee of a 15-month-old infant girl. Flexion contracture of the knee was found to be 40 degrees. An arthroscopic view showed a large and thick voluminous mediopatellar plica. It was trapped between the patella and the medial femoral condyle and it was in tight contact with the medial facet of the patella at 40 degrees flexion in the knee, blocking further extension. A longitudinal groove was noted on the articular surface of the medial femoral condyle that looked as if it had another trochlea on arthroscopic view. The mediopatellar plica came into contact with the groove at 60 degrees flexion in the knee and it fitted precisely into the groove at further flexion. The pathological plica was resected arthroscopically, which resulted in approximately 10 degrees improvement in extension of the knee. Histological examination found hypertrophy and chronic nonspecific inflammation of the synovium. The patient was helped with range-of-motion exercise and quadriceps-strengthening exercise. At 27 months follow-up, the knee had gained full extension. This article reports that a pathological mediopatellar plica may develop in infants.