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Background: The fabella is a sesamoid bone situated within the lateral head of the gastrocnemius tendon, close to the lateral femoral condyle, and adjoined to the fabellofibular ligament. It is a normal variant, found in up to 87% of patients. Fabella Syndrome describes traditionally posterolateral knee pain, occurring due to biomechanical pressure of the fabella against the lateral femoral condyle. Given its rarity, its diagnosis is often overlooked. We present a case of Fabella Syndrome with a modified surgical excision technique and review of the literature., Methods and Results: A thirty-four-year-old man presented with posterolateral knee pain following de-rotation surgery to correct a femoral malunion, from a previous femoral shaft fracture. Due to the patient's complex orthopaedic history, Fabella Syndrome was not initially diagnosed. Fabellectomy eliminated all symptoms of knee pain, with no limitations in knee function., Conclusion: Review of the literature identified ten publications (evidence level IV) describing Fabella Syndrome. This is the first reported case of Fabella Syndrome secondary to femoral de-rotation surgery. The authors recommend fabellectomy as a definitive treatment for Fabella Syndrome, in keeping with published literature.