| Summary: | Abstract Pyogenic arthritis of the atlantoaxial joint is scarce. It can lead to several complications, such as spinal cord compression and cerebral vein thrombosis. A 51‐year‐old man presented with a twenty‐day history of inflammatory neck pain. Physical examination revealed paravertebral muscle contracture, restricted neck movement, and fever. Spine magnetic resonance imaging (MRI) showed synovitis of atlanto‐odontoid joint, anterior epidural collection, and cerebral vein thrombosis. Transthoracic echocardiography was unremarkable. The patient was successfully treated with anti‐staphylococcal antibiotic treatment for 12 weeks associated with immobilization of the cervical spine. MRI performed one month after the initiation of the treatment showed disappearance of the epidural collection. The diagnosis of septic arthritis of the atlantoaxial joint should be considered in a patient with inflammatory neck pain. MRI findings are relevant in making the diagnosis of a septic atlanto‐odontoid joint. Conservative treatment, including antibiotic and neck immobilization, can be sufficient for the treatment of pyogenic arthritis of the atlantoaxial joint. Cerebral vein thrombosis is a rare complication due to septic arthritis of the atlantoaxial joint.
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