TY - JOUR
T1 - Spinal arteriovenous fistula coexisting within a spinal lipoma
T2 - report of two cases
AU - Horiuchi, Yosuke
AU - Iwanami, Akio
AU - Akiyama, Takenori
AU - Hikata, Tomohiro
AU - Watanabe, Kota
AU - Yagi, Mitsuru
AU - Fujita, Nobuyuki
AU - Okada, Eijiro
AU - Nagoshi, Narihito
AU - Tsuji, Osahiko
AU - Ishii, Ken
AU - Yoshida, Kazunari
AU - Nakamura, Masaya
AU - Matsumoto, Morio
N1 - Publisher Copyright:
© The Author(s) 2017.
PY - 2017
Y1 - 2017
N2 - Introduction 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 presentation 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. Discussion 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.
AB - Introduction 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 presentation 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. Discussion 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.
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U2 - 10.1038/S41394-017-0011-1
DO - 10.1038/S41394-017-0011-1
M3 - Article
AN - SCOPUS:85070440841
SN - 1362-4393
VL - 3
SP - 1
EP - 7
JO - Spinal Cord
JF - Spinal Cord
IS - 1
M1 - 17079
ER -