Department of Neurology, National Institute of Mental Health and Neurosciences, Bengaluru, India.
Department of Neurology, National Institute of Mental Health and Neurosciences, Bengaluru, India; Department of Clinical Neurosciences, National Institute of Mental Health and Neurosciences, Bengaluru, India.
World Neurosurg. 2021 Feb;146:e1287-e1292. doi: 10.1016/j.wneu.2020.11.153. Epub 2020 Dec 4.
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.
尽管寰枢关节不稳定在文献中很少报道,但它可与颈性肌张力障碍(cervical dystonia,CD)相关,并可能导致颅颈交界区脊髓受压。我们报告了 4 例长期 CD 伴神经并发症的病例系列。讨论了治疗策略和挑战。
回顾性分析 4 例长期 CD 伴有脊髓病或神经根病并发症的病例。
并发症平均发病年龄为 28 岁(范围 17-37 岁)。CD 的平均病程为 23.75 年。3 例患者存在颅颈交界区狭窄,其中 2 例存在齿状突游离,1 例存在寰枢关节旋转性错位。1 例患者 C3-4 水平的椎间盘干燥、膨出,脊髓内出现信号改变。药物治疗效果不佳,但肉毒毒素对所有患者均部分有效。1 例患者有脊髓病后遗症,经深部脑刺激后部分恢复。2 例行脊柱融合固定手术的患者中,1 例改善,另 1 例因脊髓不可逆损伤无改善。仅 2 例患者的总体预后良好。
早发型 CD 可导致年轻患者颈椎更高节段和颅颈交界区的脊髓并发症。多学科团队的综合管理对于早期预防并发症至关重要。