Background:Ankylosing spondylitis (AS) is a seronegative spondyloarthropathy within the spectrum of rheumatologic diseases. The systemic inflammation that characterizes AS leads to bone resorption and reformation. Pathologic remodeling may include kyphosis, osteoporosis, and multi-segment auto-fusion. Cervical fractures account for 53–78% of spinal trauma seen with AS. Surgical planning is often challenging owing to spinal deformity, medical comorbidities, the cervicothoracic foci of injury, and gross instability of these fracture.Case Description:A 55-year-old male with AS was presented with a three-column injury at the C6 level. The C6 vertebra was fractured, minimally displaced, and there was a focal kyphotic deformity. Attempted posterior fixation 2 days after presentation was aborted; the patient could not tolerate prone positioning, and there were further technical limitations to a posterior approach. Cervicothoracic fixation from C2 to T2 was then performed using the right lateral decubitus position employing the Mayfield head holder, a beanbag, and spinal neuronavigation.Conclusion:In this study, we presented a unique approach to posterior fixation of an unstable cervicothoracic fracture in a patient with AS utilizing the lateral position and neural navigation under intraoperative physiological monitoring.