• Ali Karadag, Evren Sandal, Erik H Middlebrooks, and Mehmet Senoglu.
• Department of Neurosurgery, Saglik Bilimleri University, Tepecik Research and Training Hospital, Izmir, Turkey.
• World Neurosurg. 2018 May 1; 113: 308-311.

BackgroundOchronotic arthropathy related to alkaptonuria is a rare condition. Cervical spine involvement with myelopathic features has been even more rarely described, particularly related to atlantoaxial instability. As such, little is known about the optimal surgical management in these patients.Case DescriptionWe described the first case, to our knowledge, of a patient with alkaptonuria and related cervical spondylotic myelopathy from pannus formation at the atlantoaxial joint. We describe our choices in surgical management of this rare condition in a patient with an excellent outcome.ConclusionOchronotic cervical spondylotic myelopathy is a rare condition and may require additional considerations in surgical treatment compared to more common causes of cervical spondylotic myelopathy. In our case, we elected for decompression with posterior occipitocervical screw fixation and were able to achieve neurologic recovery with no complications, currently at 2-year follow-up.Copyright © 2018 Elsevier Inc. All rights reserved.

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