Chin on chest deformity caused by upper cervical kyphosis associated with ankylosing spondylitis is rare. A 66-year-old woman presented at our institute with chief complaints of difficulty in horizontal gaze and opening her mouth. Cervical radiographs showed a C0-2 angle of 1° on flexion and 7° on extension, and her chin-brow vertical angle was 49°. We planned fixation surgery at C0-5 posteriorly to prevent the progression of kyphosis, with slight correction of the kyphosis at C0-2. The correction was performed by pushing down the over lordotically contoured titanium rods connected to an occipital plate onto the C3-5 lateral mass screws, just like cantilever technique. No palpable cracking or loss of resistance was noticed during the correction. However, intraoperative radiographs revealed apparent anterior separation of the vertebral bodies between C3 and C4. Postoperative computed tomography images at the C3/4 level suggested hemorrhage from the fracture site. Tracheostomy was performed because of massive edema around the pharynx. To secure solid bone fusion, staged surgery to extend the fusion to T3 and to graft an additional iliac bone was performed. Fortunately, the C2-7 angle was corrected to 40°, and her chin-brow vertical angle was restored to 17° without any catastrophic complications. Although the patient finally obtained an ideal sagittal alignment, the surgeon should be aware that the technique had a higher perioperative risk for iatrogenic fracture, resulting in neurological and vascular injuries.