Rotatory Subluxation and Facet Deformity in the Atlanto-occipital Joint in Patients with Chronic Atlantoaxial Rotatory Fixation: Two Case Reports

INTRODUCTION: The relationship between the morphometry of atlantoaxial joint and chronic atlantoaxial rotatory fixation (AARF) is well known, but disorders in the atlanto-occipital joint in chronic AARF are not fully elucidated. The authors report two rare cases of secondary deformities in the atlanto-occipital joint in chronic AARF. CASE REPORTS: Two patients with chronic AARF were treated with closed manipulation, skull traction and halo-vest immobilization. Clinical outcomes and radiographs were reviewed retrospectively, focusing on pathological changes in the atlanto-occipital joint using multiplanar reconstruction computed tomography and three-dimensional computed tomography images. Case 1 (12-year-old girl) had rotatory subluxation with a superior facet deformity of the atlas in the atlanto-occipital joint before the initiation of treatment. After a series of conservative treatments, both atlantoaxial and atlanto-occipital rotatory fixation could not be reduced, and both the superior facet deformity of the atlas and osteoarthritic changes in the atlanto-occipital developed. She was monitored without surgery because the disorder progressed to the spontaneous fusion of the occipital bone to the axis. Case 2 (13-year-old boy) had rotatory subluxation without facet deformity of the atlas in the atlanto-occipital joint before the initiation of treatment. However, both the superior facet deformity of the atlas and osteoarthritic changes in the atlanto-occipital developed over time, and both atlantoaxial and atlanto-occipital rotatory fixation could not be reduced after a series of conservative treatments. He still had severe neck pain and severely restricted neck mobility and underwent spinal fusion. CONCLUSION: Atlanto-occipital rotatory subluxation and facet deformity in the atlanto-occipital joint may occur after prolonged AARF. It is necessary to pay attention to pathological changes not only in the atlantoaxial joint but also in the atlanto-occipital joint, when orthopedic surgeons treat patients with AARF.