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The authors report a very rare case of marked segmental stenosis of the atlantoaxial junction causing cervical myelopathy. A 53-year-old woman suffered from burning paresthesia of both upper extremities and progressive gait disturbance for 3 years. Magnetic resonance images showed that spinal cord was markedly compressed due to hypertrophied and calcified ligamentum flavum at the atlantoaxial junction. She made a full recovery following decompressive laminectomy of atlas and axis. A posterior decompression may constitute adequate treatment and provide neurological improvement. Key words:Ligament flavum;Calcification;Atlantoaxial junction;Myelopathy;Laminectomy