Atlanto-occipital dislocation (AOD) is rarely seen in clinic because it is characteristically immediately fatal. With recent progress in the pre-hospital care, an increasing number of AOD survivors have been reported. However, because the pathophysiology of AOD is not clearly understood yet, the appropriate strategy for the initial management remains still unclear. We report a case of successful AOD treatment and describe important points in the management of this condition. It is important to note that abducens nerve palsy is a warning sign of AOD and that AOD can result in a life-threatening distortion of the arteries and the brain stem. We recommend the application of a halo vest to protect the patient's neural and vascular competence as the immediate initial step in the treatment of AOD. Horn's grading system is useful in assessing indications for surgery. Finally, when performing posterior fixation, C2 should be included because of the anatomy of the ligamentous architecture.
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