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We reported a rare characteristic anomaly of the laminae of the axis with hypoplasia of the posterior arch of atlas. A multiplane reconstruction of the computed tomography images was very necessary for treatment of this case. Possible causes of this anomaly may be the failure of ossification or fusion of the embryological term, whereas invagination of the osteophyte may be associated with the traction of the dense fibrous band during growth and development. Surgical removal of the laminae could result in a satisfactory outcome.