A large retro-odontoid cystic mass caused by transverse ligament degeneration with atlantoaxial subluxation leading to granuloma formation and chronic recurrent microbleeding case report

A large retro-odontoid cystic mass caused by transverse ligament degeneration with atlantoaxial subluxation leading to granuloma formation and chronic recurrent microbleeding case report
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DOI:
10.1016/j.spinee.2011.11.007
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发表时间:
2011-12-01
期刊:
影响因子:
4.5
通讯作者:
Takayasu, Msakazu
Takayasu, Msakazu
中科院分区:
医学2区
文献类型:
--
作者:
Takeuchi, Mikinobu;Yasuda, Muneyoshi;Takayasu, Msakazu

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背景情况:非感染性非肿瘤性齿状突后肿块是罕见的,并且没有广泛压迫脊髓的报道。我们遇到了一个大的后齿状突病变,广泛压缩脊髓。病例报告:一个76岁的男子报告说,经历了突然发作的颈部疼痛,手和脚感觉异常,构音障碍,吞咽困难。当通过外部稳定和卧床休息治疗10天后症状仍未缓解时,他被转诊到我院。颈椎动态X线片显示寰齿间距从伸展时的2 mm增宽到屈曲时的7 mm。计算机断层扫描没有发现异常的齿状突或存在的高密度区,可能会建议钙化内或附近的囊性肿块。液体衰减反转恢复轴向磁共振图像显示一个肿块,宽3.0厘米,高2.7厘米,厚2.5厘米,严重压迫下脑干。T2加权磁共振成像显示肿块包含C2致密区后方的实性部分,向吻侧延伸,与变性和肥大的韧带的存在相一致。部分囊性肿块位于硬膜外,有黄色素内容物,提示微出血。构音障碍和吞咽困难立即消失,神经系统症状消失1个月。在1年随访时,患者保持无症状,计算机断层扫描未显示肿块复发。病理诊断退行性韧带组织慢性复发微出血和相关granulationwered.Discussion:一个可能的解释,为什么囊肿生长到一个非常大的尺寸是,横韧带的轴成为退化和肥大,因为慢性机械应力寰枢椎半脱位。然后,韧带的一部分出现了反应性肉芽组织,并形成了小血管。最后,这些小血管的破裂引起反复发作的微出血,导致形成大囊肿。观察到退化的韧带组织、肉芽形成和微出血,将其与滑膜囊肿或腱鞘囊肿相鉴别。含铁血黄素沉积提示慢性复发性微出血。综合我们所有的发现,我们相信我们的病例是不同于其他已报道的病例。轴向不稳定性可能会导致大质量,如我们在这里所描述的,因此仔细观察是很重要的。(C)2011 Elsevier Inc. All rights reserved.
BACKGROUND CONTEXT: Noninfectious nontumorous retro-odontoid masses are rare, and masses have not been reported to extensively compress the spinal cord. We encountered a case of a large retro-odontoid lesion that extensively compressed the spinal cord.CASE REPORT: A 76-year-old-man reported experiencing a sudden onset of neck pain, hand and foot paresthesia, dysarthria, and dysphagia. When symptoms had not eased by 10 days of treatment with external stabilization and bed rest, he was referred to our hospital. Dynamic radiographs of the cervical spine showed that the atlantodental interval widened from 2 mm on extension to 7 mm on flexion. Computed tomography did not reveal abnormality of the odontoid process or the presence of a high-density area that could suggest calcification in or near the cystic mass. Fluid-attenuated inversion recovery axial magnetic resonance image showed a mass that was 3.0-cm wide, 2.7-cm high, and 2.5-cm thick that severely compressed the lower brain stem. T2-weighted magnetic resonance imaging showed that the mass contained a solid part posterior to the C2 dense area, extending rostrally, compatible with the presence of degenerated and hypertrophic ligaments.We performed surgical decompression of the lesion combined with atlantoaxial fixation. The partly cystic mass, which was located extradurally, had xanthochromic content, indicating microbleeding. Dysarthria and dysphagia immediately disappeared, and neurologic symptoms disappeared by 1 month. At 1-year follow-up, the patient remained symptom free, and computed tomography scans did not show recurrence of the mass. The pathologic diagnosis of degenerative ligament tissue with chronic recurrent microbleeding and associated granulation was made.DISCUSSION: A possible explanation why the cyst grew to an exceptionally large size is that the transverse ligament of axis became degenerated and hypertrophic because of chronic mechanical stress by atlantoaxial subluxation. Then, a part of the ligament developed reactive granulation with small vessel formation. Finally, rupture of these small vessels caused repeated episodes of microbleeding, resulting in formation of a large cyst. The observation of degenerative ligament tissue, granulation formation, and microbleeding differentiated it from a synovial cyst or a ganglion cyst. The presence of hemosiderin deposits suggested chronic recurrent microbleeding. Taking all our findings together, we believe that our case of retro-odontoid cystic mass is different from the others that have been reported. Atlantoaxial instability may cause a large mass, such as we described here, so that careful observation is important. (C) 2011 Elsevier Inc. All rights reserved.