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Esophageal perforation in ankylosing spondylitis (AS) is a rare complication in anterior cervical spine surgery and has not been reported before. A 50-year-old patient with AS developed incomplete tetraplegia after minimal trauma. C5 pedicle fracture was diagnosed and treated predominantly by physical therapy until neurological symptoms progressed. Cervical spine MRI showed C6/7 fracture and spinal cord compression. The patient underwent dorsal laminectomy, C5-7 anterior cervical fusion using allograft iliac crest and CASPAR-plate fixation. Delayed esophageal perforation appeared 10 months postoperatively when he came first to our hospital. He complained of dysphagia and developed acute dyspnea. Posterior stabilization with two plates was performed followed by removal of the ventral plate and screws. The esophageal laceration was sutured. The patient was treated with antibiotics and percutaneous endoscopic gastrostomy. Position of fracture and implants were accurate at 18 months postoperatively. The patient had persistent minor neurological deficits (Frankel D) at last follow-up. We conclude that esophageal perforation after anterior spinal fusion is a rare complication. Minor traumas in patients with AS are unstable and can result in significant spinal injury. Dorsoventral stabilization should be performed to avoid further complications.