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NI FEATURE: FACING ADVERSITY…TOMORROW IS ANOTHER DAY! - LETTER TO EDITOR
Year : 2017  |  Volume : 65  |  Issue : 2  |  Page : 380-382

Massive epistaxis resulting from radiation-induced internal carotid artery pseudoaneurysm


1 Department of Neurosurgery, Rush University Medical Center, Chicago, Illinois, USA
2 Department of Neurosurgery, Rush University Medical Center, Chicago, Illinois; Department of Neurosurgery, University Hospitals Cleveland Medical Center, Case Western Reserve University School of Medicine, Cleveland, Ohio, USA

Date of Web Publication10-Mar-2017

Correspondence Address:
Manish K Kasliwal
Department of Neurosurgery, University Hospitals Cleveland Medical Center, Case Western Reserve University School of Medicine, 11100 Euclid Avenue, Mailstop HAN 5042, Cleveland, Ohio – 44106
USA
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/neuroindia.NI_1259_15

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How to cite this article:
Nazari P, Tan LA, Wewel JT, Moftakhar R, Kasliwal MK. Massive epistaxis resulting from radiation-induced internal carotid artery pseudoaneurysm. Neurol India 2017;65:380-2

How to cite this URL:
Nazari P, Tan LA, Wewel JT, Moftakhar R, Kasliwal MK. Massive epistaxis resulting from radiation-induced internal carotid artery pseudoaneurysm. Neurol India [serial online] 2017 [cited 2017 May 23];65:380-2. Available from: http://www.neurologyindia.com/text.asp?2017/65/2/380/201832


Sir,

Massive epistaxis is generally due to hypertension, with trauma and vascular abnormalities representing less than 5% of the cases. Sudden and massive epistaxis after radiotherapy is usually fatal, and it is one of the most dreaded complications among patients with cancer of the head and neck region.[1],[2],[3],[4] With the increasing involvement and popularity of endovascular techniques in the field of neurosurgery, neurosurgeons are often consulted for such clinical presentations and their management. We describe a case of massive epistaxis due to rupture of radiation-induced internal carotid artery (ICA) pseudoaneurysm in a patient with nasopharyngeal carcinoma (NPC) and present a comprehensive review of the literature regarding the clinical characteristics and different management options for radiation-induced internal carotid artery (ICA) pseudoaneurysm.

A 52-year-old man with significant past medical history of NPC presented to our Emergency Department with acute worsening of epistaxis and syncope. He was treated in 2004 with chemotherapy and external beam radiation therapy (EBRT). Following a recurrence after 10 years of initial treatment, he underwent another episode of radiation treatment. Following this treatment, he reported recurrent episodes of epistaxis at home, occurring approximately twice a week over several months prior to his presentation to our hospital. Computed tomographic (CT) scan of the brain and skull base showed no intracranial changes. During his admission to the hospital, he had 2 episodes of massive epistaxis with significant blood loss (~400–500cc) for which anterior and posterior nasal packing was performed by the otolaryngologists. Considering tumor sources for possible vascular erosion or aneurysm formation,, a CT angiography of the brain and neck was performed to evaluate the source of bleeding that was suspicious of arising from a pseudoaneurysm [Figure 1]. A conventional angiogram was subsequently performed to further elucidate the anatomy of the pseudoaneurysm and its possible embolization to control bleeding. Anteroposterior and lateral angiograms confirmed the presence of a 4.7 × 4.7 × 2.9 mm fusiform pseudoaneurysm just proximal to the cervical/petrous segment junction of the left ICA with vigorous venous drainage into large dilated varices directed to the posterior nasopharynx. The left ICA was completely coil embolized [Figure 2]. Post-procedure CT perfusion showed development of infarcts involving the left temporal and parietal lobes with corresponding changes on perfusion. The patient was discharged to a rehabilitation center; however, he remained aphasic (Broca's type) at his last follow up at 12 months.
Figure 1: Sagittal view of CT angiogram of the brain and neck demonstrating a bulbous dilatation of the distal cervical left internal carotid artery (white arrow) adjacent to the skull base predominantly along its ventral aspect, indicating the presence of a pseudoaneurysm

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Figure 2: Anteroposterior view of the cerebral angiogram showing a large pseudoaneurysm forming a fistula with a large venous structure (black arrow)

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The formation of an ICA pseudoaneurysm is one of the uncommon complications seen after radiation treatment of NPC and typically occurs at the petrous and laceral segemnts of the ICA, which may present with life-threatening epistaxis. Patients with a second course of radiotherapy have a higher risk of radiation damage to the surrounding tissue and are more prone to skull base osteoradionecrosis.[5],[6],[7] The mechanism of radiation-induced vascular injury is not fully understood, but may result from obstruction of the vasa vasorum, premature atherosclerosis, adventitial fibrosis, and necrosis of the arterial wall. Combined with high blood pressure of the vessel, it can result in the rupture of the arterial wall and even dissection with extravasation of blood.[6],[8],[9],[10],[11] As subclinical infection from osteonecrosis of the skull base is also implicated in the development of ICA pseudoaneurysm, there are increased chances of development of ICA pseudoaneurysm following a second course of radiation, which is also associated with increased chances of osteonecrosis of the skull base. The development of newer modalities of radiation, such as intensity modulated radiotherapy (IMRT), may lead to decreased chances of radiation-induced complications, including the development of a pseudoaneurysm, by decreasing the radiation exposure to the nearby vital structures. Even in patients, who have undergone EBRT when diagnosed with NPC for the first time, IMRT or any other conformal modality should be employed for re-radiation to decrease the likelihood of this dreaded vascular complication, which is reported to occur more frequently after the second dose of radiation.

Review of the literature revealed very few articles reporting the development of ICA pseudoaneurysm following radiation for NPC, most being case reports and a few small case series. The relevant details of the cases reported are described in [Table 1]. As can be clearly seen, endovascular treatment remains the preferred modality of treatment. The study by Lam et al.,[7] reported that successful temporary control of bleeding and airway are the two statistically significant factors, which predicted survival in this group of patients. In fact, their study showed that one-third of the patients died from failure to control bleeding or from the complications of treatment, which emphasizes the dreaded nature of this complication and underscores the early involvement of both otolaryngologists and neurosurgeons/interventional neuroradiologists. Even in patients who survived the treatment, approximately 16% suffered from severe neurological complications, as was seen in our patient. The complications were mostly ischemic in nature arising due to stent occlusion and thromboembolism.[1],[5],[6],[7],[8],[9],[14],[15],[16],[17],[18],[19],[20]
Table 1: Review of the treatment modality used for various radiation induced ICA pseudoaneurysms in the literature

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The key to improving the overall outcome in patients who develop ICA pseudoaneurysm and who subsequently become symptomatic due to epistaxis is to recognize the etiology and carry out a multimodality management. The importance of past medicalhistory, especially any history of radiation, is important to uncover this rare complication. In patients with massive epistaxis, it is important to consult otolaryngologists early because the control of symptomatic bleeding may often involve anterior or posterior nasal packing. A CTA can provide a clue to the existence of an ICA pseudoaneurysm. However, conventional angiography remains the key to the definitive diagnosis of this condition and for formulating treatment. Recognized treatments for these lesions include the use of cellulose acetate polymer, detachable balloons, wall stents, endovascular coils, and surgical ligation. While the ideal goal of treatment is to occlude the aneurysm while preserving the parent vessel, sometimes, it is difficult to achieve this aim even with current endovascular techniques, which are the first modalities of treatment, due to the weakened vascular structures and the surrounding radionecrotic bone; or, because the morphology of these lesions may not be suitable for coiling.[5],[9],[18],[12],[13],[21] Sacrifice of the carotid artery is the last option under such circumstances and can be performed when there is demonstration of adequate collateral circulation via the circle of Willis, as evaluated using the angiogram. Nevertheless, sacrifice of the carotid artery has a high risk of cerebrovascular morbidity and mortality. Our patient probably developed thromboembolism from the pseudoaneurysm, which resulted in a relatively poor outcome. If the patient cannot tolerate the balloon test occlusion test, an extracranial-to-intracranial bypass surgery should be contemplated. 5–22% of patients passing the balloon occlusion test may still develop ischemic complications, including the development of a cerebral infarct.[3],[6],[16],[17],[19]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 » References Top

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Crow WN, Scott BA, Guinto FC Jr, Chaljub G, Wright G, Rabassa AE,et al. Massive epistaxis due to pseudoaneurysm. Treated with detachable balloons. Arch Otolaryngol Head Neck Surg 1992;118:321-4.  Back to cited text no. 4
    
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Lam JW, Chan JY, Lui WM, Ho WK, Lee R, Tsang RK. Management of pseudoaneurysms of the internal carotid artery in postirradiated nasopharyngeal carcinoma patients. Laryngoscope 2014;124:2292-6.  Back to cited text no. 7
    
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Chen HC, Lin CJ, Jen YM, Juan CJ, Hsueh CJ, Lee JC, et al. Ruptured internal carotid pseudoaneurysm in a nasopharyngeal carcinoma patient with skull base osteoradionecrosis. Otolaryngol Head Neck Surg 2004;130:388-90.  Back to cited text no. 8
    
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Cheng KY, Lee KW, Chiang FY, Ho KY, Kuo WR. Rupture of radiation-induced internal carotid artery pseudoaneurysm in a patient with nasopharyngeal carcinoma: Spontaneous occlusion of carotid artery due to long-term embolizing performance. Head Neck 2008;30:1132-5.  Back to cited text no. 9
    
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