Ankylosing spondylitis (AS) is a chronic inflammatory rheumatological disease affecting the axial skeleton with various extra-articular complications. Dysphagia due to a giant anterior osteophyte of the cervical spine in AS is extremely rare. We present a 48-year-old male with AS suffering from progressive dysphagia to soft foods and liquids. Esophagography showed an anterior osteophyte at C5-C6 resulting in esophageal compression. The patient refused surgical resection of the osteophyte and received conservative therapy. However, after 6 months there was no improvement in dysphagia. This case illustrates that a large cervical osteophyte may be the cause of dysphagia in patients with AS and should be included in the diagnostic workup in early stages of the disease. © 2013 The Korean Association of Internal Medicine.