Dysphagia after occipital cervical fusion for retro-odontoid pseudotumor with ossification of the anterior longitudinal ligament

Abstract

Background: Ossification of the anterior longitudinal ligament (OALL) of the cervical spine is a relatively rare disease. If patients present with dysphagia, hoarseness, and/or dyspnea, they may require surgery.

Case description: Over a 7-month period, a 55-year-old female with a history of cerebral palsy developed a progressive quadriparesis accompanied by diffuse sensory loss (i.e., clumsiness of the hand/legs and gait disturbance). The cervical spine X-rays showed atlanto-axial subluxation with instability, while the cervical MRI demonstrated "pseudotumor in the retro-odontoid" region. Following an occipital cervical fusion (C0-C2) surgery, her quadriparesis resolved. Nevertheless, she had persistent dysphagia that worsened over 6 months. Video fluoroscopy revealed severe mechanical stenosis of the pharynx, which was attributed to OALL extending from the C3-C6 levels. Following OALL resection through a right anterior approach utilizing diamond burrs and an ultrasonic bone curette, the dysphagia rapidly resolved.

Conclusion: We report a rare case of retro-odontoid pseudotumor successfully treated with a posterior C0-C2 cervical fusion. Additional symptomatic C3-C6 OALL, responsible for progressive dysphagia, was later managed with focal anterior OALL resection.

Keywords: Atlanto-axial subluxation; Dysphasia; Occipital cervical fusion; Symptomatic OALL; Video fluoroscopy.

Figure 1:

Preoperative MRI showed severe compression of spinal nerves due to retro-odontoid tumor (a: T2WI, b: T1WI). Cervical X-ray showed OALL like a bird’s beak at the C3-6 level (c), and follow-up MRI 6 months after the first surgery clearly showed that the cerebrospinal fluid around the spinal cord had recovered and the retro-odontoid tumor was shrinking (d).

Figure 2:

Aggressive OALL is prominent on cervical X-ray after the first surgery (a). Preoperative video fluoroscopy revealed a filling defect due to OALL at the C5/6 level (b,c). The red arrow in Figure 2b indicates contrast loss. Cervical X-ray after OALL resection showing adequate smoothening of the OALL (d). In the operative field, the OALL is flattened with a diamond burr (e).