A case of ankylosing hyperostosis of the cervical spine is reported because of the rarity of the condition. A man aged 59 years was first seen on April 15, 1981 with the chief complaint of neck pain. Rentogenograms showed ankylosing hyperostoses of the cervical spine at the level of C-2 to C-7. On January 12, 1985, he complained of hard dysphagia and hoarseness. Rentgenograms showed the development of hyperostosis at the level of C-2 to C-4. The disturbance of passage of contrast medium was also observed at the level of C-3 to C-4 in esophagogram. On January 18, 1985, anterior surgery was porformed in which the central bony mass was removed. The lateral bone mass was left in place so that the stability of cervical spine remained too. This procedure will disturb reossification. Complete relief was obtained immediately after surgery and he has been asymptomatic 9 months postoperatively.