Case Report

Onyx Embolization of a Ruptured Rotundum Foreman Artery Aneurysm in a Patient with Moyamoya Disease: A Case Report Peng Liu, Xianli Lv, Youxiang Li, Ming Lv

Key words Aneurysm - Embolization - Moyamoya disease - Onyx - Rotundum foreman -

Abbreviations and Acronyms MMD: Moyamoya disease Department of Interventional Neuroradiology, Beijing Neurosurgical Institute and Beijing Tiantan Hospital, Capital Medical University, China To whom correspondence should be addressed: Youxiang Li, M.D. [E-mail: [email protected]] Citation: World Neurosurg. (2015) 84, 4:1178.e1-1178.e3. http://dx.doi.org/10.1016/j.wneu.2015.06.039 Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2015 Elsevier Inc. All rights reserved.

INTRODUCTION Approximately 3e14% of patients with Moyamoya disease (MMD) are accompanied by cerebral aneurysm (14). In a retrospective analysis of 111 MMD patients with 131 aneurysms by Kawaguchi et al. (6), the distribution of aneurysms in MMD locating around circle of Willis, in the basal ganglia, and on the collateral vessels was 3:1:1, respectively. Aneurysms in MMD usually are classified into 2 major types (6). One is the major cerebral artery aneurysm (true aneurysm), which is defined as outpouching sac caused by dilation of all 3 intact layers of the artery. The histology of the true aneurysm is an inflammation disease with aneurysm wall involving a multitude of inflammatory cells and mediators in previous studies (2, 5). The other type is the peripheral artery aneurysm, which usually is a pseudoaneurysm because of its appearance on cerebral angiograms (12, 13, 17), with a fibrous wall involving all layers of the artery in communication with a parent vessel. Rupture of this type of aneurysm usually is associated with intrac ranial hemorrhage or intraventricular

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- BACKGROUND:

Hemorrhage caused by the rupture of a rotundum foreman artery pseudoaneurysm in Moyamoya disease (MMD) is rarely reported.

- CASE

DESCRIPTION: We report a case of 46-year-old man with MMD presenting with left temporal lobe hemorrhage. Cerebral angiogram showed a pseudoaneurysm located on the rotundum branch of the left internal maxillary artery, which anastomosed with a pial vessel. Using onyx 18, we successfully embolized the pseudoaneurysm. The patient was discharged neurologically intact.

- CONCLUSION:

Given the difficulties and risks of surgery, onyx embolization of rotundum foreman artery aneurysm accompanied with MMD is feasible and effective.

hemorrhage. In addition, it is deemed that rupture of the peripheral artery aneurysm is caused by vessel wall stress induced by stenosis of anterior circulation (6). The rotundum foreman artery, a terminal branch of internal maxillary artery, is a very rare location for peripheral artery aneurysm in MMD. We described the first case of a male MMD patient with a rotundum foreman artery aneurysm presenting with intracerebral hemorrhage.

CASE REPORT A 46-year-old man presented with a sudden-onset headache followed by unconsciousness for 4 hours. A computed tomography scan revealed a hematoma in the left temporal lobe (Figure 1). The patient was responsive to pain, the left pupil was fixed dilated, and there was a right Babinski response. The removal of the left temporal lobe hematoma and decompressive craniotomy was performed, and the patient was awake the following day (Figure 2). One month later, contrastenhanced magnetic resonance imaging was performed to seek the cause for hemorrhage and showed a suspicious aneurysm located at left middle cranial fossa (Figure 3A and B). The subsequent digital subtraction angiography manifested a bilateral Moyamoya vascular pattern (Figure 4A and B) and confirmed a

pseudoaneurysm arising from the rotundum branch of left internal maxillary vessel corresponding to the magnetic resonance image (Figure 4C and D). Emission computed tomography perfusion images revealed that hemodynamic status of the bilateral hemispheres were decreased (Figure 3C). After a detailed discussion, we decided to perform endovascular treatment for the patient. Under general anesthesia, a 6F Guiding catheter (Envoy; Codman Neuro, Colorado Springs, Colorado, USA) was positioned in the distal left external carotid artery, through which a Marathon Flow Directed Micro Catheter (ev3 Endovascular, Inc., Plymouth, Minnesota, USA) was advanced over a microguidewire (Traxcess-14; MicroVention, Tustin, California, USA) into the rotundum artery via the internal maxillary artery. Superselective contrast injection demonstrated a 3.7-mm aneurysm. Moreover, the parent artery communicated with the intracranial artery (Figure 4E). Given the potential existence of dangerous collaterals, under constant fluoroscopic visualization, 0.5 mL of Onyx 18 was infused slowly into the aneurysm without reflux. Immediate postoperative angiography demonstrated the disappearance of the pseudoaneurysm (Figure 4F). Twelve days later, because of the damage of the distal branch of the superficial

WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2015.06.039

CASE REPORT PENG LIU ET AL.

RUPTURED ROTUNDUM FOREMAN ARTERY ANEURYSM TREATED WITH ONYX

Figure 3. One month later, axial (A) and coronal (B) contrast-enhanced magnetic resonance imaging showed a suspicious aneurysm located at the left middle cranial fossa (arrow). The 99mTc emission computed tomography showed the decreased perfusion of the bilateral hemispheres were decreased (C). Figure 1. Computed tomography scan showed a hemotoma in the left temporal lobe.

temporal artery during the previous decompressive craniotomy surgery (Figure 4D), the left encephalo-duroarterio-synangiosis was not planed, and the left cranioplasty was performed. Three months later, we performed right encephalo-duro-arterio-synangiosis for the patient. There was no neurologic deterioration when the patient was discharged from hospital. The patient suffered no rebleeding events during the 2-year followup. Informed consent was obtained from the individual participant included in the study to publish related materials. Institutional review board approval was obtained from Beijing Tiantan Hospital’s ethics committee for this study.

Figure 2. On the next day after the hematoma removal and decompressive craniotomy, computed tomography showed that the hematoma was entirely cleared.

DISCUSSION To our knowledge, there is no literature in which authors report an internal maxillary artery aneurysm associated with MMD. There are only 36 published cases reports or series of aneurysms of the maxillary artery system not associated with MMD (3, 4, 11, 15, 16). In the

majority of these reports, the aneurysmal enlargement of the maxillary artery is false or pseudoaneurysmal in nature with or without histology verification. The etiology of these aneurysms were partial arterial wall injury, potential complication after radiation therapy (4, 15), and iatrogenic causes (surgery, biopsy,

Figure 4. The bilateral internal carotid artery angiograms confirmed the diagnosis of Moyamoya disease, right (A) and left (B). The anteroposterior (C) and lateral (D) angiogram of the left external carotid showed a pseudoaneurysm of rotundum branch of the internal maxillary artery and the damage of the distal branch of the superficial temporal artery during the prior decompressive craniotomy surgery (short arrows). The superselective angiogram showed the pseudoaneurysm was associated with a pial vessel (D). The postprocedural lateral angiogram of the left external carotid artery showed the disappearance of pseudoaneurysm (E). Immediate post-operative angiography demonstrated the disappearance of the pseudo-aneurysm (F).

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catheterization, and fine-needle aspiration) (3, 11, 16). In our case, the rotundum branch of the internal maxillary artery served as a collateral for intracranial circulation. The natural history of peripheral aneurysms in MMD is still unknown. Previous studies explained that the course of these aneurysms are the remaining Moyamoya vessels caused by previous rupture of friable vessels, and this aneurysmal lesion is the direct cause of rebleeding in MMD, suggesting that hemodynamic stress is involved in the development of such aneurysms (6, 7). Patients with aneurysms of this type may suffer frequent, short-term rebleeding, and rapid expansion; urgent and reasonable treatment is needed. However, some reports suggest an aneurysm of this type may spontaneously regress during the observation period (4, 12, 13). In the treatment of peripheral aneurysms associated with MMD, several factors should be taken into consideration, including the location of the aneurysm, supplying territory, as well as the potential anastomosis with other vessels. The major concern is the potential dangerous anastomosis with other vessels, such as the dangerous anastomosis with the lateral branch of cavernous segment of internal carotid artery must be kept in mind. With the development of endovascular materials and techniques, the number of peripheral aneurysms in MMD treated with Onyx and N-butyl cyanoacrylate is increasing (1, 7-10). Embolization has the advantage of being able to reach vessels beyond the limitations of surgery and has lower reported complication rates. Coil embolization was not an option in our case because of the very small caliber of the vessel. Onyx, an embolic agent, has been used widely in the treatment of peripheral aneurysms (8-10). Because of its nonadhesive nature, Onyx allows longer, slower, and more controlled injections with precise delivery of the embolic material. Because of its lower viscosity compared with Onyx 34, Onyx 18 has the

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RUPTURED ROTUNDUM FOREMAN ARTERY ANEURYSM TREATED WITH ONYX

ability to travel more distally along the internal maximal artery and was therefore the best suited to our case. On the basis of the current advances in endovascular techniques, the positive results reported with endovascular therapy recently and the obvious risks of surgery, we recommend treating the rotundum foreman artery aneurysms with endovascular embolization, especially in patients with MMD. For other neurosurgeons encountering similar circumstances, we believe our experience may be helpful in choosing treatment options. REFERENCES 1. Choulakian A, Drazin D, Alexander MJ: NBCA embolization of a ruptured intraventricular distal anterior choroidal artery aneurysm in a patient with moyamoya disease. J Neurointerv Surg 2: 368-370, 2010. 2. Chyatte D, Bruno G, Desai S, Todor DR: Inflammation and intracranial aneurysms. Neurosurgery 45:1137-1146; discussion 1146-1147, 1999. 3. Duwe KM, Newhouse JH, Fayter J, Stern L, Budorick NE: Conservative management of an extrarenal pseudoaneurysm after percutaneous needle biopsy of a renal allograft. J Ultrasound Med 19:281-283, 2000. 4. Ernemann U, Herrmann C, Plontke S, Schafer J, Plasswilm L, Skalej M: Pseudoaneurysm of the superior thyroid artery following radiotherapy for hypopharyngeal cancer. Ann Otol Rhinol Laryngol 112:188-190, 2003. 5. Frosen J, Piippo A, Paetau A, Kangasniemi M, Niemela M, Hernesniemi J, Jääskeläinen J: Remodeling of saccular cerebral artery aneurysm wall is associated with rupture: histological analysis of 24 unruptured and 42 ruptured cases. Stroke 35:2287-2293, 2004. 6. Kawaguchi S, Sakaki T, Morimoto T, Kakizaki T, Kamada K: Characteristics of intracranial aneurysms associated with moyamoya disease. A review of 111 cases. Acta Neurochir (Wien) 138: 1287-1294, 1996. 7. Kim SH, Kwon OK, Jung CK, Kang HS, Oh CW, Han MH, Kim YS, Baik SK: Endovascular treatment of ruptured aneurysms or pseudoaneurysms on the collateral vessels in patients with moyamoya disease. Neurosurgery 65:1000-1004; discussion 1004, 2009. 8. Lv X, Jiang C, Li Y, Yang X, Wu Z: Endovascular treatment for cerebral perforating artery aneurysms. Neurol Res 33:553-557, 2011.

9. Lv X, Li Y, Liu A, Zhang J, Wu Z: Parent artery occlusion for peripheral anterior inferior cerebellar artery aneurysm. A case report and review of the literature. Neuroradiol J 21:261-265, 2008. 10. Lv X, Wu Z, Li Y, Yang X, Jiang C, Sun Y, Zhang N: Endovascular treatment of cerebral aneurysms associated with arteriovenous malformations. Eur J Radiol 81:1296-1298, 2012. 11. McNamara MP Jr, Boden T: Pseudoaneurysm of the breast related to 18-gauge core biopsy: successful repair using sonographically guided thrombin injection. AJR Am J Roentgenol 179: 924-926, 2002. 12. Ni W, Xu F, Xu B, Liao Y, Gu Y, Song D: Disappearance of aneurysms associated with moyamoya disease after STA-MCA anastomosis with encephaloduro myosynangiosis. J Clin Neurosci 19: 485-487, 2012. 13. Satoh T, Yamamoto Y, Asari S, Sakurai M, Suzuki K: Disappearance and development of cerebral aneurysms in moyamoya disease. Case report. J Neurosurg 58:949-953, 1983. 14. Takahashi JC, Miyamoto S: Moyamoya disease: recent progress and outlook. Neurol Med Chir (Tokyo) 50:824-832, 2010. 15. Walker AT, Chaloupka JC, Putman CM, Abrahams JJ, Ross DA: Sentinel transoral hemorrhage from a pseudoaneurysm of the internal maxillary artery: a complication of CT-guided biopsy of the masticator space. AJNR Am J Neuroradiol 17:377-381, 1996. 16. Welch TJ, Sheedy PF 2nd, Johnson CD, Johnson CM, Stephens DH: CT-guided biopsy: prospective analysis of 1,000 procedures. Radiology 171:493-496, 1989. 17. Yuasa H, Tokito S, Izumi K, Hirabayashi K: Cerebrovascular moyamoya disease associated with an intracranial pseudoaneurysm. Case report. J Neurosurg 56:131-134, 1982.

Conflict of interest statement: Supported by the Commission of Beijing Municipal Science and technology, municipal clinical special application study, special fund project (No. Z14110000211441). Received 11 February 2015; accepted 16 June 2015 Citation: World Neurosurg. (2015) 84, 4:1178.e1-1178.e3. http://dx.doi.org/10.1016/j.wneu.2015.06.039 Journal homepage: www.WORLDNEUROSURGERY.org Available online: www.sciencedirect.com 1878-8750/$ - see front matter ª 2015 Elsevier Inc. All rights reserved.

WORLD NEUROSURGERY, http://dx.doi.org/10.1016/j.wneu.2015.06.039

Onyx Embolization of a Ruptured Rotundum Foreman Artery Aneurysm in a Patient with Moyamoya Disease: A Case Report.

Hemorrhage caused by the rupture of a rotundum foreman artery pseudoaneurysm in Moyamoya disease (MMD) is rarely reported...
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