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Klippel-Feil syndrome is characterized by congenital fusion of cervical vertebrae with a wide range of associated anomaly. The authors present a 50-year-old Klippel-Feil syndrome patient with a minor trauma followed progressive quadriparesis. He had typical radiologic findings of type II Klippel-Feil syndrome and presented progressive myelopathy due to cord compression at foramen magnum level with cervical instability. The patient underwent craniocervical decompression and fusion. The authors reviewed the pertinent literatures and discussed this rare syndrome. Key words:Klippel-Feil syndrome;Myelopathy;Spinal instability;Occipitocervical fusion.