Cervical Myeloradiculopathy and Atlantoaxial Instability in Cervical Dystonia

Atlantoaxial instability, although rarely reported in the literature, can be associated with cervical dystonia (CD) and may lead to compression of the cord at the craniovertebral junction. We present a case series of 4 patients of longstanding CD with neurologic complications. Treatment strategies a...

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Bibliographic Details
Published in:World neurosurgery 2021-02, Vol.146, p.e1287-e1292
Main Authors: Neeraja, Koti, Prasad, Shweta, Surisetti, Bharath Kumar, Holla, Vikram Venkappayya, Sharma, Dinesh, Kamble, Nitish, Kulanthaivelu, Karthik, Dwarakanth, Srinivas, Pruthi, Nupur, Pal, Pramod Kumar, Yadav, Ravi
Format: Article
Language:English
Subjects:
Acetylcholine Release Inhibitors - therapeutic use
Adolescent
Adult
Atlanto-Axial Joint - physiopathology
Atlanto-Axial Joint - surgery
Atlantoaxial instability
Botulinum Toxins - therapeutic use
Cervical dystonia
Cord compression
Craniovertebral junction
Deep Brain Stimulation
Female
Humans
Intervertebral Disc Degeneration - etiology
Intervertebral Disc Degeneration - physiopathology
Intervertebral Disc Degeneration - surgery
Joint Instability - etiology
Joint Instability - physiopathology
Joint Instability - therapy
Male
Nerve Block
Os odontoideum
Radiculopathy - etiology
Radiculopathy - physiopathology
Radiculopathy - therapy
Spinal Cord Compression - etiology
Spinal Cord Compression - physiopathology
Spinal Cord Compression - therapy
Spinal Fusion
Torticollis - complications
Torticollis - physiopathology
Torticollis - therapy
Young Adult
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Summary:Atlantoaxial instability, although rarely reported in the literature, can be associated with cervical dystonia (CD) and may lead to compression of the cord at the craniovertebral junction. We present a case series of 4 patients of longstanding CD with neurologic complications. Treatment strategies and challenges are discussed. Retrospective analysis of 4 cases of longstanding CD with complications of myelopathy or radiculopathy. The average age at onset of complications was 28 years (range, 17–37). The average duration of CD was 23.75 years. Narrowing of the craniovertebral junction was seen in 3 patients, of which 2 had os odontoideum, and 1 had rotational malalignment at the atlantoaxial joint. One patient had disc desiccation with bulge and intramedullary signal changes in the cord at C3-4 level. Medical treatment was not satisfactory, but botulinum toxin was partly useful in all. One patient had sequelae of myelopathy and did recover partially after deep brain stimulation. Of the 2 patients who underwent surgical fixation with a fusion of the spine, one improved, and the other had no improvement due to irreversible cord damage. The overall outcome was satisfactory only in 2 patients. Early-onset CD can lead to cord complications at a young age and at higher levels of the cervical spine and at the cervicovertebral junction. Comprehensive management by a multidisciplinary team is crucial to prevent complications early.
ISSN:1878-8750
1878-8769
DOI:10.1016/j.wneu.2020.11.153