Maki Yoshinori, Ishibashi Ryota, Fukuda Hitoshi, Kobayashi Miyako, Chin Masaki, Yamagata Sen
Department of Neurosurgery, Kurashiki Central Hospital.
Neurol Med Chir (Tokyo). 2018 Apr 15;58(4):185-188. doi: 10.2176/nmc.cr.2017-0241. Epub 2018 Feb 23.
Vertebral arteriovenous fistula (VAVF), which can cause subarachnoid hemorrhage (SAH) when having a perimedurally drainage, has been reported as a rare vascular abnormality in patients with neurofibromatosis type 1 (NF-1). In addition, extracranial vertebral aneurysm (EVAn) coexisting with VAVF and NF-1 is considered rare, and further complication with SAH is extremely rare in patients. There is only one reported case of NF-1 complicated with SAH from VAVF with an EVAn. Here, we present a case of a middle-aged patient with NF-1. The VAVF accompanied by an EVAn was detected with an episode of SAH. The VAVF with an EVAn in our case was accompanied with an epidural varix, lacking of perimedullary drainage, which could be a cause for SAH. We speculate the mechanism of SAH from the VAVF with an EVAn lacking of perimedurally drainage, focusing on hemodynamic stress of the VAVF and the tissue fragility related to NF-1.
椎动脉动静脉瘘（VAVF）在经硬脊膜周围引流时可导致蛛网膜下腔出血（SAH），据报道，这是1型神经纤维瘤病（NF-1）患者中一种罕见的血管异常。此外，与VAVF和NF-1共存的颅外椎动脉动脉瘤（EVAn）被认为很罕见，并且患者发生SAH的进一步并发症极为罕见。仅有1例关于NF-1合并VAVF伴EVAn导致SAH的报道。在此，我们报告1例中年NF-1患者。该患者在发生SAH时被检测出伴有EVAn的VAVF。我们病例中的伴有EVAn的VAVF伴有硬膜外静脉曲张，缺乏硬脊膜周围引流，这可能是SAH的一个原因。我们推测缺乏硬脊膜周围引流的伴有EVAn的VAVF导致SAH的机制，重点关注VAVF的血流动力学压力以及与NF-1相关的组织脆弱性。
