Introduction: Proximal tibiofibular joint osteoarthritis is a rare disease most commonly occurring in the presence of either systemic inflammatory conditions or severe knee osteoarthritis. Case Presentation: The authors present a case report of isolated bilateral tibiofibular arthrosis in an otherwise healthy 28-year-old female patient. The patient presented with complaints of lateral knee pain. Radiographs and magnetic resonance imaging (MRI) revealed bilateral tibiofibular joint osteoarthritis. Trauma, repetitive use injury, and rheumatologic causes were excluded. The patient’s pain was initially managed conservatively, but she subsequently underwent tibiofibular arthrodesis for progressive pain symptoms. We review in this report the current literature on tibiofibular joint osteoarthritis and its treatment. Conclusion: Isolated tibiofibular joint osteoarthritis is a rare condition that may progress and requires surgical treatment to achieve optimal clinical outcomes.