Rationale: Kashin–Beck disease (KBD) is known for some typical characters like finger joint enlargement, shortened fingers, and dwarfism. However, Avascular necrosis (AVN) of the talus in KBD has rarely been reported in the literature. Here, we reported on a KBD patient presented with partial AVN of the talus in conjunction with ankle and subtalar arthritis. Patient concerns: A 50-year-old woman presented with severe pain and limited range of motion in her left ankle and subtalar joint while walking for 2 years. She had been walking with the aid of crutches for many years. Conservative treatment with rigid orthosis and activity restriction could not help reduce the pain in the left foot. Diagnoses: Radiographs demonstrated that partial AVN was developed in the body of the talus and arthritis was viewed in the left ankle and subtalar joint. Hence, we established the diagnosis of partial talar AVN in conjunction with ankle and subtalar arthritis. Interventions: A conservative tibiotalocalcaneal fusion attempting to preserve as much viable talar body as possible was performed using a humeral locking plate and 2 cannulated compression screws. Outcomes: Bone union proved by CT scan and a good alignment of the left limb were achieved at 4-month follow-up postoperatively. Lessons: Partial AVN of the talus along with ankle and subtalar arthritis in KBD patients has rarely been reported as it is not a common characteristic of KBD in clinical practice. Conservative tibiotalocalcaneal fusion could help preserving much more viable talar body, maintaining most structural integrity of the ankle joint, and achieving a stable and plantigrade foot postoperatively.

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