Horiuchi Yosuke, Iwanami Akio, Akiyama Takenori, Hikata Tomohiro, Watanabe Kota, Yagi Mitsuru, Fujita Nobuyuki, Okada Eijiro, Nagoshi Narihito, Tsuji Osahiko, Ishii Ken, Yoshida Kazunari, Nakamura Masaya, Matsumoto Morio
1Department of Orthopedic Surgery, Keio University School of Medicine, Tokyo, Japan.
4Present Address: Department of Orthopedic Surgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku, Tokyo, 160-8582 Japan.
Spinal Cord Ser Cases. 2017 Nov 14;3:17079. doi: 10.1038/s41394-017-0011-1. eCollection 2017.
Spinal lipoma and spinal arteriovenous fistula (sAVF) are different pathologies and their co-existence is extremely rare. Here we reported two cases of adult-onset sAVF occurring within a spinal lipoma and with review the literature in an attempt to identify the mechanisim of and optimal treatment of this condition.
Case 1 was a 51-year-old man who was treated by embolization of the feeding artery and ligation of the draining vein. Case 2 was a 53-year-old man who was treated by embolization and resection of the tumor containing the shunt zone. In both cases, symptoms improved after surgery. However, in Case 1, angiography at 1 month after the surgery revealed recurrence of the arteriovenous shunt.
A literature search revealed only nine other similar case reports. All cases, including ours occurred in adults. In almost all cases, the shunt was located within the spinal lipoma. Pathologic examination revealed venous hypertension, but no evidence of congenital vascular malformation. Given that lipomas release angiogenic factors, the presence of a spinal lipoma may indicate its involvement in the development of acquired sAVF. Our two cases might represent a new subtype of sAVF. Based on our experiences, we recommend resection of the tumor containing the shunt for the management of sAVF.
脊髓脂肪瘤和脊髓动静脉瘘(sAVF)是不同的病理情况,它们同时存在极为罕见。在此,我们报告了两例成年发病的sAVF发生于脊髓脂肪瘤内的病例,并回顾文献以试图明确这种情况的机制及最佳治疗方法。
病例1是一名51岁男性,接受了供血动脉栓塞和引流静脉结扎治疗。病例2是一名53岁男性,接受了包含分流区域的肿瘤栓塞和切除术。两例患者术后症状均有改善。然而,病例1术后1个月的血管造影显示动静脉分流复发。
文献检索仅发现另外9例类似病例报告。所有病例,包括我们的病例,均发生于成年人。几乎所有病例中,分流均位于脊髓脂肪瘤内。病理检查显示静脉高压,但无先天性血管畸形的证据。鉴于脂肪瘤释放血管生成因子,脊髓脂肪瘤的存在可能表明其参与了后天性sAVF的发生发展。我们的两例病例可能代表了sAVF的一种新亚型。根据我们的经验,我们建议切除包含分流的肿瘤以治疗sAVF。