Atlantoaxial instability: An exceptional complication of ankylosing spondylitis

Background: Atlantoaxial instability (AAI) and more rarely odontoid pannus formation, similar to the one observed in Rheumatoid Arthritis, are seldom reported in ankylosing spondylitis (AS). We report a new case of a patient with AS with a pannus in the atlanto-axial region and cervical C1-C2 instability. Case presentation: The patient, now aged 41, was diagnosed with AS in 2010. She was put on different non steroidal anti inflammatory treatments with persistent spinal pain. She was referred to our department in 2015 with severe cervical pain and stiffness since 3 months. On examination, the patient had severely limited cervical spine movements. Lumbar spine movements were moderately affected. There was no neurological deficit. Her Bath Ankylosing Spondylitis Disease Activity Index (BASDAI) was 4.9/10 and Bath Ankylosing Spondylitis Functional Index (BASFI) was 5/10. Plain Radiographs of the pelvis showed bilateral grade 4 sacroilitis and bilateral coxitis. Dorsal and lumbar spine plain radiographs showed squaring of vertebral bodies. Cervical spine radiographs showed an increased atlanto-axial distance. Spinal MRI confirmed the atlantoaxial subluxation with an anterior distance of 8 mm, with marked intraspinal pannus formation and synovitis around the odontoid peg. Synovial thickening exerted an anterior mark on the bulbo-medullary junction with no evidence of oedema signs. Posterior zygapophysial ankylosis involving all cervical levels was also observed. The patient had a cervical collar with anti-tumor necrosis factor-α (cerolizumab-pegol) prescription with favorable outcome. Conclusion: Odontoid pannus formation is rare in AS. Clinical and radiological follow-up are important to assess the impact on the cervical spine. Keywords: Ankylosing spondylitis, Atlantoaxial instability, MRI, Cerolizumab-pego