- Department of Neurosurgery, Nippon Medical School Chiba Hokusoh Hospital, 1715 Kamagari, Inzai, Chiba, Japan
- Department of Neurosurgery, Nippon Medical School Hospital, 1-1-5, Sendagi, Bunkyoku, Tokyo, Japan
Department of Neurosurgery, Nippon Medical School Hospital, 1-1-5, Sendagi, Bunkyoku, Tokyo, Japan
DOI:10.4103/2152-7806.141887Copyright: © 2014 Suzuki M. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
How to cite this article: Suzuki M, Umeoka K, Kominami S, Morita A. Successful treatment of a ruptured flow-related aneurysm in a patient with hemangioblastoma: Case report and review of literature. Surg Neurol Int 26-Sep-2014;5:
How to cite this URL: Suzuki M, Umeoka K, Kominami S, Morita A. Successful treatment of a ruptured flow-related aneurysm in a patient with hemangioblastoma: Case report and review of literature. Surg Neurol Int 26-Sep-2014;5:. Available from: http://sni.wpengine.com/surgicalint_articles/successful-treatment-of-a-ruptured-flow-related-aneurysm-in-a-patient-with-hemangioblastoma-case-report-and-review-of-literature/
Background:No cerebral aneurysms on the feeder associated with hemangioblastomas that ruptured before resection have been reported. We report a patient with a ruptured flow-related aneurysm associated with cerebellar hemangioblastoma and a tumor feeder treated simultaneously by a single procedure of embolization using N-butyl cyanoacrylate before tumor removal.
Case Description:A 36-year-old female with a cerebellar tumor was admitted to our institute. Four days later, she suffered a massive subarachnoid hemorrhage mainly in the posterior fossa. Left vertebral angiograms showed an aneurysm on the feeding artery, posterior inferior cerebellar artery. Both the aneurysm and its main feeder were simultaneously treated by a single procedure of embolization using N-butyl cyanoacrylate. Their complete obliteration was confirmed angiographically. Four days after the procedure, we removed the tumor and the embolized aneurysm. The pathological diagnosis was hemangioblastoma and flow-related ruptured aneurysm.
Conclusion:Cerebral angiography should be performed to rule out vascular abnormalities such as cerebral aneurysms adjacent to the tumor in patients with hemangioblastoma who present with intracranial hemorrhage. We emphasize the usefulness of embolization with N-butyl cyanoacrylate for hemangioblastoma with ruptured feeder aneurysm, by which the aneurysm and the feeder could be simultaneously embolized.
Keywords: Cerebral aneurysm, embolization, hemangioblastoma, N-butyl cyanoacrylate, subarachnoid hemorrhage
Cerebral aneurysms associated with hemangioblastomas are very rare[
A 36-year-old healthy female was admitted to our hospital due to the sudden onset of a severe headache. Gadolinium-enhanced magnetic resonance imaging (MRI) revealed an enhanced mass in the cerebellar vermis [
Left vertebral angiograms. (a)Intraoperative view before embolization (b) Superselective angiograms from the microcatheter show a dumbbell-shaped aneurysm just distal to the catheter tip. (c) NBCA injection into the aneurysm and the distal and proximal part of the feeder. (d) Complete disappearance of the aneurysm and the anterior part of the tumor stain seen in
(a) Intraoperative findings. Note the ruptured aneurysm (arrow) on the feeder near the vascular-rich tumor (asterisk). (b) Microphotograph of the aneurysm showing disruption of the internal elastic lamina and rupture of the adventitia (Elastica van Giesson staining, original magnification ×10)
Glacker et al.[
Although many cases about hemorrhage associated with hemangioblastomas have been reported in patients without other vascular anomalies, only six studies[
Cerebral angiographs of intracranial hemangioblastomas typically show intense tumor blush from the feeding arteries and enlargement of the drainers in the late arterial- or capillary phase. These findings are indicative of high tumor vascularity and an increase in the regional blood flow from a slightly enlarged feeding artery to the draining vein. This results in hemodynamic stress that may lead to the formation of an aneurysm on the feeder as like brain arteriovenous malformations (AVMs). In previous large studies of brain AVMs, the incidence of flow-related aneurysms associated with AVMs ranged from 10.9% to 30.7%, and from 45% to 83.3% in patients who manifested hemorrhage.[
Whether flow-related aneurysms are at increased risk for rupture and whether they should be addressed before the treatment of AVMs remains controversial.[
The preoperative embolization of hemangioblastomas has been debated.[
We encountered a patient with hemangioblastoma and a ruptured aneurysm on the distal PICA, the main feeder. The flow-related aneurysm was treated by embolization using NBCA and the hemangioblastoma was subsequently resected totally. Merit of this treatment is that the aneurysm and the tumor feeder are simultaneously embolized by a single procedure. Cerebral angiographs should be obtained to rule out vascular anomalies near the tumor and to avoid unpredictable intraoperative bleeding in patients with hemangioblastoma and hemorrhage.
1. Cornelius JF, Saint-Maurice JP, Bresson D, George B, Houdart E. Hemorrhage after particle embolization of hemangioblastomas: Comparison of outcomes in spinal and cerebellar lesions. J Neurosurg. 2007. 106: 994-8
2. de San Pedro JR, Rodriguez FA, Niguez BF, Sanchez JF, Lopez-Guerrero AL, Murcia MF. Massive hemorrhage in hemangioblastomas Literature review. Neurosurg Rev. 2010. 33: 11-26
3. Glasker S, Van Velthoven V. Risk of hemorrhage in hemangioblastoma of the central nervous system. Neurosurgery. 2005. 57: 71-6
4. Guzman R, Grady MS. An intracranial aneurysm on the feeding artery of a cerebellar hemangioblastoma. Case report. J Neurosurg. 1999. 91: 136-8
5. Lv X, Wu Z, Li Y, Jiang C, Yang X, Zhang J. Cerebral arteriovenous malformations associated with flow-related and circle of willis aneurysms. World Neurosurg. 2011. 76: 455-8
6. Meisel HJ, Mansmann U, Alvarez H, Rodesch G, Brock M, Lasjaunias P. Cerebral arteriovenous malformations and associated aneurysms: Analysis of 305 cases from a series of 662 patients. Neurosurgery. 2000. 46: 793-800
7. Menovsky T, Andre Grotenhuis J, Bartels RH. Aneurysm on the anterior inferior cerebellar artery (AICA) associated with high-flow lesion: Report of two cases and review of literature. J Clin Neurosci. 2002. 9: 207-11
8. Murai Y, Kobayashi S, Tateyama K, Teramoto A. Persistent primitive trigeminal artery aneurysm associated with cerebellar hemangioblastoma. Case report. Neurol Med Chir (Tokyo). 2006. 46: 143-6
9. Murai Y, Kominami S, Yoshida Y, Mizunari T, Adachi K, Koketsu K. Preoperative liquid embolization of cerebellar hemangioblastomas using N-butyl cyanoacrylate. Neuroradiology. 2012. 54: 981-8
10. Redekop G, TerBrugge K, Montanera W, Willinsky R. Arterial aneurysms associated with cerebral arteriovenous malformations: Classifications, incidence, and risk of hemorrhage. J Neurosurg. 1998. 89: 539-46
11. Sakamoto N, Ishikawa E, Nakai Y, Akutsu H, Yamamoto T, Nakai K. Preoperative endovascular embolization for hemangioblastoma in the posterior fossa. Neurol Med Chir (Tokyo). 2012. 52: 878-84
12. Schmidt NO, Reitz M, Raimund F, Treszi A, Grzyska U, Westphal M. Clinical relevance of associated aneurysms with arteriovenous malformations of the posterior fossa. Acta Neurochir Suppl. 2011. 112: 131-5
13. Ueno K, Mabuchi S, Echizenya K, Isu T, Goto S. Incidentally-discovered aneurysm – report of eight cases. No Shinkei Geka. 1977. 5: 183-8
14. Westphal M, Grzyska U. Clinical significance of pedicle aneurysms on feeding vessels, especially those located in infratentorial arteriovenous malformations. J Neurosurg. 2000. 92: 995-1001
15. Yoshii Y, Maki Y, Tomono Y, Nakamura T. Cerebellar hemangioblastoma with multiple aneurysms. No To Shinkei. 1976. 28: 703-8
16. Zager EL, Shaver EG, Hurst RW, Flamm ES. Distal anterior inferior cerebellar artery aneurysms. Report of four cases. J Neurosurg. 2002. 97: 692-6