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脊柱旁动静脉分流(PAVS)的临床特征和治疗策略:一项系统评价和个体参与者数据荟萃分析。

Clinical features and treatment strategy of paraspinal arteriovenous shunt (PAVS): a systematic review with individual participants data meta-analysis.

机构信息

Department of Neurosurgery, Armed Forces Capital Hospital, Gyeonggi-do, Republic of Korea.

Department of Neurosurgery, Sooncheonhyang Seoul University Hospital, Seoul, Republic of Korea.

出版信息

Eur Spine J. 2021 Aug;30(8):2385-2400. doi: 10.1007/s00586-021-06831-2. Epub 2021 Apr 23.

Abstract

BACKGROUND

Because of the rarity of the disease, paraspinal arteriovenous shunt (PAVS) is not well recognized, and therapeutic options remain controversial. To introduce a rare disease of PAVS and demonstrate its etiology, clinical features, treatment options, and outcomes, we presented a case report and conducted a systematic review and individual participants data (IPD) meta-analysis.

METHODS

Studies regarding on PAVS were integrated and IPD were obtained including patients' demographics, disease etiology, clinical and radiologic features, clinical courses and outcomes. Clinical manifestation and treatment outcomes were reviewed, and comparison analysis (cervical versus thoracolumbar) were performed. Further, logistic regression analyses were conducted to identify the poor prognostic factors (incomplete obliteration).

RESULTS

Fifty-two articles were selected, and 88 patients enrolled. General and location-specific characteristics of PAVSs were identified: '3/4 of the isolated and 1/4 of the associated etiology', 'bruit, thrill, or murmur (cervical) and weakness (thoracolumbar) as common symptoms', '40% multiple feeders', and '22% intradural venous involvement'. Endovascular treatment was usually preferred (75%). Of 88 enrolled patients, 18 patients showed incomplete obliteration (20.5%). In multivariate analysis, 'etiologies of systematic genetic dysplasia (P = 0.031) and trauma (negatively, 0.038)' were significantly associated with incomplete obliteration. The parameters of 'multiple feeders (0.066)' and 'combined approach (negatively, 0.065)' are verified only in univariate analysis.

CONCLUSION

General as well as location-specific characteristics of PAVS is successfully demonstrated. Approximately 20% of the incomplete obliteration is noted, and three potential poor prognostic factors are identified, namely, 'etiology of systematic genetic dysplasia (positive) and trauma (negative)', 'combined approach (negative), and 'multiple feeders'.

摘要

背景

由于该病罕见,因此对脊柱旁动静脉分流(PAVS)认识不足,治疗方案仍存在争议。为了介绍一种罕见的 PAVS 疾病,并展示其病因、临床特征、治疗选择和结果,我们报告了一例病例并进行了系统回顾和个体参与者数据(IPD)荟萃分析。

方法

综合了有关 PAVS 的研究,并获得了包括患者人口统计学、疾病病因、临床和影像学特征、临床病程和结局的 IPD。回顾了临床表现和治疗结果,并进行了比较分析(颈椎与胸腰椎)。此外,还进行了逻辑回归分析,以确定不良预后因素(不完全闭塞)。

结果

选择了 52 篇文章,并纳入了 88 例患者。确定了孤立性和相关性 PAVS 的一般和特定部位特征:“3/4 为孤立性,1/4 为相关性病因”、“颈椎常见症状为杂音、震颤或杂音,胸腰椎常见症状为无力”、“40%为多个供血者”和“22%为硬脊膜内静脉受累”。通常首选血管内治疗(75%)。在 88 例纳入的患者中,有 18 例出现不完全闭塞(20.5%)。在多变量分析中,“系统性遗传发育不良(P=0.031)和创伤(负相关,0.038)的病因”与不完全闭塞显著相关。“多个供血者(0.066)”和“联合治疗(负相关,0.065)”的参数仅在单变量分析中得到验证。

结论

成功地展示了 PAVS 的一般和特定部位特征。注意到约 20%的不完全闭塞,并确定了三个潜在的不良预后因素,即“系统性遗传发育不良(阳性)和创伤(阴性)病因”、“联合治疗(阴性)”和“多个供血者”。

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