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Dural ossification (DO) is a degenerative phenomenon of endorhachis characterized by the displacement of dura by proliferative osseous tissue, which is a common complication of spinal ligament ossifications. Precisely preoperative diagnosis and elaborate surgical manipulation are the focus of clinicians since incorrect management of DO during surgery could induce cerebrospinal fluid leakage (CSF) and spinal cord injury (SCI). However, there has been no case of postoperative DO which was reported in ankylosing spondylitis (AS). Herein, we reported a 49-year-old AS patient who presented delayed incomplete paraplegia due to the development of extensive thoracolumbar DO after L3 pedicle subtraction osteotomy (PSO), aiming to help spinal surgeons to have a more comprehensive understanding of the possible pathogenic mechanism, diagnosis, surgical and prognostic implications of DO in AS patients.