Pyogenic Atlantoaxial Rotational Dislocation Representing Adult Torticollis with Vertebral Artery Occlusion: A Case Report and Review.

Abstract

Pyogenic atlantoaxial rotational dislocation is a rare but life-threatening condition. Disease progression damages the soft tissue and bony structure, resulting in spinal cord or brain stem compression. Rapid and correct diagnosis could enable successful treatment. However, few studies have been reported, and the characteristics of a successful treatment course are not well known. In addition, our case presented with vertebral artery (VA) occlusion. Here we present a report of successful treatment of pyogenic atlantoaxial rotational dislocation representing adult torticollis with VA occlusion. A 67-year-old woman with neck pain and high fever was treated for suspected meningitis. Although her fever improved, pain persisted for several weeks. Examinations mostly showed characteristics of an infection with destructive atlantoaxial rotational dislocation; however, positive uptake with positron emission tomography, no anomalies with diffusion-weighted magnetic resonance imaging, and VA occlusion indicated the presence of a tumor lesion. After VA embolization and Halo reduction/stabilization, biopsy and blood culture revealed pyogenic infection. Antibiotics with rigid stabilization improved the inflammation and allowed definitive occipitocervical fixation, resulting in an uneventful postoperative course and painless bony fusion 2 years postoperatively. VA involvement should be examined for safe treatment. Stabilization is warranted to improve chronic inflammation. Knowledge regarding the characteristic treatment course could enable successful treatment planning and may prove to be lifesaving.