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脊髓动静脉节段性综合征(SAMS)中硬膜外高流量血管畸形的自发消退:一例独特病例报告

Spontaneous regression of extradural high-flow vascular malformation in spinal arteriovenous metameric syndrome (SAMS): A unique case report.

作者信息

Iampreechakul Prasert, Chuntaroj Songpol, Wattanasen Yodkhwan, Hangsapruek Sunisa, Lertbutsayanukul Punjama, Siriwimonmas Somkiet

机构信息

Department of Neurosurgery, Neurological Institute of Thailand, Bangkok, Thailand.

Department of Neuroradiology, Neurological Institute of Thailand, Bangkok, Thailand.

出版信息

Surg Neurol Int. 2023 May 5;14:163. doi: 10.25259/SNI_4_2023. eCollection 2023.

Abstract

BACKGROUND

Spinal arteriovenous metameric syndrome (SAMS) is a rare nonhereditary genetic vascular disorder, involving multiple layers of tissues in the same metameric level. Spontaneous regression of SAMS has never been reported in the medical literature.

CASE DESCRIPTION

A 42-year-old woman presented with intermittent low back pain for 6 months. Magnetic resonance imaging of the thoracolumbar spine incidentally found clusters of spinal vascular malformations involving spinal cord, vertebral bodies, epidural space, and paraspinal muscles. There was no sign of venous congestion. Magnetic resonance angiography and spinal angiography revealed intradural spinal cord arteriovenous malformation (SCAVM) at the level of T10-11 and extradural high-flow osseous arteriovenous fistula. Due to asymptomatic SAMS and a high risk of anterior spinal arterial compromise during treatment, conservative treatment was considered in our patient. Spinal angiography obtained 8 years after initial angiography demonstrated significant regression of extradural component of SAMS and stable intradural SCAVM.

CONCLUSION

We describe a unique case of SAMS with spontaneous regression of extradural component during a long-term observation period.

摘要

背景

脊髓动静脉节段性综合征(SAMS)是一种罕见的非遗传性基因血管疾病,累及同一节段水平的多层组织。医学文献中从未报道过SAMS的自发消退情况。

病例描述

一名42岁女性出现间歇性腰痛6个月。胸腰椎磁共振成像偶然发现累及脊髓、椎体、硬膜外间隙和椎旁肌肉的脊髓血管畸形簇。无静脉淤血迹象。磁共振血管造影和脊髓血管造影显示T10 - 11水平的硬脊膜内脊髓动静脉畸形(SCAVM)和硬膜外高流量骨动静脉瘘。由于患者为无症状性SAMS且治疗期间存在脊髓前动脉受损的高风险,我们的患者考虑采取保守治疗。初次血管造影8年后进行的脊髓血管造影显示SAMS的硬膜外成分显著消退,硬脊膜内SCAVM稳定。

结论

我们描述了一例独特的SAMS病例,在长期观察期间硬膜外成分出现自发消退。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2bf9/10246388/bc1f741b9b3e/SNI-14-163-g001.jpg

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