This paper reports a case of diffuse pigmented villonodular synovitis (DPVNS), associated with advanced gonarthrosis, submitted to total knee replacement. The patient had progressive pain and swelling. She had two previous surgeries, firstly arthroscopic synovectomy and subsequently open synovectomy associated with radiotherapy, with recurrence of the disease. Magnetic resonance imaging revealed diffuse synovitis, advanced arthrosis, and bone cysts. The patient was submitted to a total knee replacement and synovectomy. There was a good postoperative clinical course, with improvement of pain, function, and joint edema on examination. The patient will be followed regarding the possibility of disease recurrence and implant survival.

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