Background: Cauda equina syndrome (CES) is a rare neurologic complication in patients with long-standing ankylosing spondylitis (AS). MRI characteristically shows an enlarged caudal thecal sac, bone erosion and dural ectasia. Dural ectasia is more likely to occur in the posterior elements rather than the vertebral bodies. Methods: We report an uncommon case of CES in AS with formation of anterior dural ectasia. The origin of this rare pathology is analysed and relevant literatures are reviewed. Results: The patient had a 50-year history of AS and presented with weakness of his left leg but without bladder or rectal dysfunction. Radiographs of the full spine showed the classic features of AS with fusion of all spinal ligaments and bilateral sacroiliac joints. CT scanning demonstrated erosion-like lesions with marginal sclerosis in the posterior aspect of the T12 and L1 vertebral bodies. Lumbar MRI revealed anterior dural ectasia into the vertebral body, and ventral herniation of the spinal cord and adhesion to the dural sac. Conclusions: The clinical manifestations of anterior dural ectasia in AS with CES include lower limb numbness and weakness, bladder and rectal dysfunction, impotence, etc. MRI characteristically shows an enlarged caudal thecal sac, bone erosion and anterior dural ectasia at the junction of thoracolumbar and upper lumbar.