Dysphagia after occipital cervical fusion for retro-odontoid pseudotumor with ossification of the anterior longitudinal ligament
- Department of Neurosurgery, Kawasaki Saiwai Hospital, Kawasaki, Japan.
Hidenori Matsuoka, Department of Neurosurgery, Kawasaki Saiwai Hospital, Kawasaki, Japan.
DOI:10.25259/SNI_286_2022Copyright: © 2022 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
How to cite this article: Hidenori Matsuoka, So Ohashi, Michihisa Narikiyo, Ryo Nogami, Hirokazu Nagasaki, Yoshifumi Tsuboi. Dysphagia after occipital cervical fusion for retro-odontoid pseudotumor with ossification of the anterior longitudinal ligament. 29-Apr-2022;13:184
How to cite this URL: Hidenori Matsuoka, So Ohashi, Michihisa Narikiyo, Ryo Nogami, Hirokazu Nagasaki, Yoshifumi Tsuboi. Dysphagia after occipital cervical fusion for retro-odontoid pseudotumor with ossification of the anterior longitudinal ligament. 29-Apr-2022;13:184. Available from: https://surgicalneurologyint.com/surgicalint-articles/11560/
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
Cervical ossification of the anterior longitudinal ligament (OALL), a subtype of diffuse idiopathic skeletal hyperostosis (DISH), rarely causes dysphagia requiring direct anterior cervical resection.[
Over a 7-month period, a 55-year-old female with a history of athetoid cerebral palsy developed a progressive quadriparesis. Cervical X-rays showed atlanto-axial subluxation (AAS) with instability. The cervical MR showed a retro-odontoid “pseudotumor” focally compressing the ventral cord, while the cervical CT showed OALL extending from C3-C6. For her progressive myelopathy and retro-odontoid mass with AAS, she underwent that a C0-C2 posterior fusion with C1 was arch resection. Postoperatively, although her quadriparesis resolved, she exhibited increasing dysphagia., The follow-up MRI demonstrated regression of the retro-odontoid pseudotumor [
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).
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).
OALL, also called Forestier’s disease, is considered a subtype of DISH.[
Patients with retro-odontoid “pseudotumor” may require a posterior C0-C2 fusion. Here, the preoperative workup should include both an MR and CT to determine if there is any accompanying subaxial cervical OALL that may become symptomatic following the C0-C2 fusion and later require direct anterior OALL resection.
The authors certify that they have obtained all appropriate patient consent.
There are no conflicts of interest.
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