A high-grade pleomorphic intra-articular liposarcoma of the knee is described in a 48-year-old man, which was diagnosed histologically after arthroscopy of the knee for suspected pigmented villonodular synovitis (PVNS). The patient proceeded to undergo an extra-articular resection with a custom-made prosthesis. This report highlights the need to remember this rare tumour in the differential diagnosis of atypical soft tissue lesions within the knee joint prior to instrumentation.