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Rationale: Intra-articular corticosteroid injection (IACI) is a cost-effective conservative treatment of mild-to-moderate osteoarthritis. Adverse events after this procedure range from life-threatening systemic reactions to self-limiting local reactions. To our knowledge, this is the 1st report of osteonecrosis (ON) in the medial tibial plateau after IACI. Patient concerns: An 81-year-old female visited our hospital due to left knee pain of increasing intensity. She presented the sudden onset of severe acute knee pain with long lasting knee pain for several years. Diagnosis: The diagnosis was confirmed ON of medial tibial plateau of knee joint by pathologic finding. Interventions: We conducted a posterior stabilized total-knee arthroplasty with no requirement for bone grafting or additional prosthesis, such as metal augments or stems. Outcomes: At the postoperative 1 year follow-up, the patient was satisfied with the surgery and had no pain during walking and active knee motion. Lessons: This case especially stress the possibility of ON in medial tibia plateau after IACI. Therefore, clinicians should monitor symptoms after IACI to enable early detection of this complication.