Translate this page into:
Notice: Please configure GTranslate from WP-Admin -> Settings -> GTranslate to see it in action.
Ossification of the posterior longitudinal ligament at the craniocervical junction presenting with Brown-Séquard syndrome: A case report
-
Received: ,
Accepted: ,
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, tweak, 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: Schuermans VNE, Van Aalst J, Postma AA, Smeets AYM. Ossification of the posterior longitudinal ligament at the craniocervical junction presenting with Brown-Séquard syndrome: A case report. Surg Neurol Int 2021;12:501.
Abstract
Background:
Several case reports about spinal cord compression due to hyperostosis at the craniocervical junction are available. However, compression at C1-C2 solely due to ossification of the posterior longitudinal ligament (OPLL) is rare.
Case Description:
A 50-year-old Asian male, with a history of lumbar spinal canal stenosis, presented with a progressive quadriparesis within 3 months. Imaging showed central OPLL at the C1-C2 level contributing to severe spinal cord compression. The patient improved neurologically after a C1-C2 laminectomy.
Conclusion:
A patient presented with a progressive Brown-Séquard syndrome due to OPLL at the craniocervical junction (C1-C2 level) and improved following a decompressive laminectomy.
Keywords
Brown-Séquard syndrome
Craniocervical junction
Myelopathy
Ossification of the posterior longitudinal ligament
INTRODUCTION
The literature about high cervicomedullary junction ossification of the posterior longitudinal ligament (OPLL) is limited. Here, we report a case in which OPLL contributed to severe C1-C2 spinal cord compression resulting in myelopathy/quadriparesis that largely resolved following posterior decompressive surgery.
CASE PRESENTATION
A 50-year-old male of Asian origin presented with a progressive quadriparesis of 3 months’ duration [Table 1]. He had undergone lumbar surgery for spinal stenosis at the L4L5 level in the same year. The cervical MRI and CT scans both showed C1-C2 central anterior compression due to OPLL [Figures 1 and 2].



Surgery
The patient underwent a C1-C2 laminectomy without fusion. The postoperative MR performed 6 months later confirmed adequate cord decompression [Figure 3]. Over the next 2 years, the patient’s neurological deficit largely resolved.

DISCUSSION
Stenosis of the spinal canal at the craniocervical junction due to OPLL and/or ossification of the transverse atlantal ligament is rare.[1] Certo et al. described a series of seven patients with retro-odontoid masses who were successfully treated with a C1 laminectomy and C1-C2 fixation.[2] Takemoto et al. also described a series of 10 patients with retro-odontoid pseudotumors that were similarly treated with a C1 laminectomy without instrumentation.[4] Although posterior decompression with instrumentation is generally recommended following upper cervical spine decompression to avoid instability to avoid and progressive kyphosis, in this case, a focused C1-C2 laminectomy without instrumentation sufficed.[3,5]
CONCLUSION
A patient with central C1-C2 OPLL was successfully decompressed with a partial laminectomy without instrumentation.
Ethical statement
The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
The patient has consented to submission of the case report and publication in the journal.
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013).
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent.
Financial support and sponsorship
Dr. A. Postma has received institutional grants from Siemens Healthineers and Bayer.
Conflicts of interest
There are no conflicts of interest.
REFERENCES
- Case Rep Neurol Med. 2012;2012:893284.Atlas hypoplasia and ossification of the transverse atlantal ligament: A rare cause of cervical myelopathy.
- [Google Scholar]
- Acta Neurochir Suppl. 2019;125:259-64.Retroodontoid degenerative pseudotumour causing spinal cord compression and myelopathy: Current evidence on the role of posterior C1-C2 fixation in treatment.
- [Google Scholar]
- Spine (Phila Pa 1976). 2014;39:58-67.Long-term results of cervical myelopathy due to ossification of the posterior longitudinal ligament with an occupying ratio of 60% or more.
- [Google Scholar]
- Clin Spine Surg. 2016;29:E514-21.Clinical and radiographic outcomes of C1 laminectomy without fusion in patients with cervical myelopathy that is associated with a retro-odontoid pseudotumor.
- [Google Scholar]
- Spine (Phila Pa 1976). 2019;44:E150-6.Radiological characteristics and clinical outcome of ossification of posterior longitudinal ligament involving C2 after posterior laminoplasty and instrumented fusion surgery.
- [Google Scholar]