Cerebrospinal fluid leak presented with the C1-C2 sign caused by spinal canal stenosis: a case report
Cerebrospinal fluid leak presented with the C1-C2 sign caused by spinal canal stenosis: a case report
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DOI：
10.1186/s12883-020-01697-1
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发表时间：
2020-04-23
期刊：
影响因子：
2.6
通讯作者：
Miyajima, Masakazu
中科院分区：
文献类型：
--
作者：
Akiba, Chihiro;Bandai, Hideki;Miyajima, Masakazu
研究背景颅内低血压是一种以脑脊液（CSF）丢失引起的低CSF压为特征的疾病。虽然脑脊液漏的一些机制已被阐明，椎管狭窄从未被报道为颅内低血压的病理原因。C1-C2征是一种特征性的影像学特征，它表明C1和C2棘突之间的CSF聚集，偶尔在低颅压患者的磁共振成像（MRI）上观察到。病例报告一位58岁男性，主诉颈椎后疼痛持续3个月，沿着四肢麻木和肌无力。MRI脂肪抑制T2加权像显示C1-C2节段脊髓后区积液，111 In-DTPA脑池造影明确显示脑脊液漏入同一区域。MRI还显示C3/4水平的椎管狭窄，计算机断层扫描（CT）脊髓造影显示同一水平的阻塞。我们诊断为脑脊液漏引起的低颅压，这可能是由C3/4节段椎管狭窄引起的。尽管保守治疗72小时，脑CT显示双侧硬膜下血肿的发展。因此，我们同时进行了硬膜下血肿钻孔引流，C1/2节段的血液补片治疗和C3-4节段的椎板成形术。术后症状改善，影像学表现提示脑脊液漏和硬膜下血肿。结论本病例的脑脊液漏表现为C1-C2节段脊髓后区积液。由于椎管狭窄导致硬膜内压力增加，导致硬膜撕裂。由于存在尾侧椎管狭窄以及C1-C2节段脊柱后区组织结构的脆弱性，CSF漏入硬膜外腔，随后漏入C1-C2节段的脊柱后区。因此，我们的理论支持了先前报道的与C1-C2征相关的CSF动力学机制，并且我们认为椎管狭窄是颅内压降低的一种新病因。
Background Intracranial hypotension is a disorder characterized by low cerebrospinal fluid (CSF) pressure typically caused by loss of CSF. Although some mechanisms account for the CSF leakage have been elucidated, spinal canal stenosis has never been reported as a pathological cause of intracranial hypotension. C1-C2 sign is a characteristic imaging feature, which indicates CSF collection between the spinous processes of C1 and C2, occasionally observed on magnetic resonance imaging (MRI) in patients with intracranial hypotension. Case presentation A 58-year-old man was presented to our institute with complaints of posterior cervical pain persisting for 3 months, along with numbness and muscle weakness of extremities. A fat suppression T2-weighted image of MRI illustrated fluid collection in the retrospinal region at C1-C2 level, and an 111In-DTPA cisternoscintigram clearly revealed the presence of CSF leakage into the same region. The MRI also showed stenosis in spinal canal at C3/4 level, and a computed tomography (CT) myelogram suggested a blockage at the same level. We gave a diagnosis as intracranial hypotension due to the CSF leakage, which might be caused by the spinal canal stenosis at C3/4 level. Despite 72 h of conservative therapy, a brain CT showed the development of bilateral subdural hematomas. We, therefore, performed burr-hole drainage of the subdural hematoma, blood-patch therapy at C1/2 level, and laminoplasty at C3-4 at the same time. Improvement of symptoms and imaging features which suggested the CSF leak and subdural hematoma were obtained post-operatively. Conclusion The present case suggested the mechanism where the CSF leakage was revealed as fluid collection in the retrospinal region at C1-C2 level. Increased intradural pressure due to the spinal canal stenosis resulted in dural tear. CSF leaked into the epidural space and subsequently to the retrospinal region at C1-C2 level, due to the presence of spinal canal stenosis caudally as well as the vulnerability of the tissue structure in the retrospinal region at C1-C2 level. Thus, our theory supports the mechanisms of previously reported CSF dynamics associated to C1-C2 sign, and also, we suggest spinal canal stenosis as a novel etiology of intracranial hypotension.