• Yoshinori Ishikawa, Naohisa Miyakoshi, Michio Hongo, Yuji Kasukawa, Daisuke Kudo, and Yoichi Shimada.
• Department of Orthopedic Surgery, Akita University Graduate School of Medicine, Akita, Japan. Electronic address: isikaway@doc.med.akita-u.ac.jp.
• World Neurosurg. 2020 Dec 1; 144: 82-87.

BackgroundPyogenic 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.Case DescriptionA 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.ConclusionsVA 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.Copyright © 2020 Elsevier Inc. All rights reserved.

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