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Nontraumatic and postirradiated intracavernous carotid hemorrhage:

an unusual case of epistaxis and review of the literature

Jing-Jing Wang, MDa, Yong Wang, MDb, Po-Hung Chang, MDc,Ta-Jen Lee, MDc, De-Hui Wang, MDa,

aDepartment of Otolaryngology-Head and Neck Surgery, Eye, Ear, Nose and Throat Hospital, Shanghai Medical College,

Fudan University, Shanghai, PR ChinabDepartment of Neurosurgery, Jiao Tong University, Shanghai, PR China

cDivision of Rhinology, Department of Otolaryngology, Chang Gung Memorial Hospital, Taoyuan, Taiwan

Received 9 August 2009

Abstract Intracavernous carotid hemorrhage is a rare cause of epistaxis. We present a case of epistaxis caused

by postradiotherapy and nontraumatic cavernous internal carotid artery (ICA) hemorrhage. An 80-

year-old man was admitted to our hospital with a one week history of recurrent left-sided epistaxis

and a past history of radiotherapy after radical maxillectomy. Emergent angiography revealed a leak

in the cavernous segment of the ICA and subsequent detachable balloon occlusion embolization of

the left internal carotid artery was performed without sequelae. We conclude that carotid artery

hemorrhage must be considered in the differential diagnosis of profuse and recurrent epistaxis,

especially for patients after craniofacial radiotherapy. ICA embolization is the definitive treatment

provided cross circulation is adequate.

2011 Elsevier Inc. All rights reserved.

1. Introduction

Epistaxis is the most common emergency in otorhinolar-

yngology [1]. It may result from a multitude of causes, both

local and systemic. Most cases are due to bleeding from the

anterior nasal septum and are easily managed with local

measures. Posterior epistaxis is more severe, with a distinct

source of bleeding often difficult to localize. Common

etiologic factors include mucosal dryness, digital trauma,

nasal septal deviation, anticoagulation drug in use, and

hypertension. Uncommon etiologic factor for epistaxis is

trauma that, together with vascular abnormalities, accounts

for fewer than 5% of severe cases [2-4].

Rupture of postirradiated great vessels is rare. Fewer than

10 cases have been reported in the English literature [5]. The

most common presentation for nontraumatic and postirra-

diated cavernous internal carotid artery (ICA) rupture is

pseudoaneurysm. Several cases in the literature report this

complication of craniofacial radiotherapy, all in those with

nasopharyngeal carcinoma [6-8].

Patients with epistaxis who fail initial conservative

therapy require endoscopic cautery, surgical ligation, or

transarterial embolization of the nasal cavity vascular supply.

Embolization is primarily targeted at the branches arising

from the internal maxillary artery [4]. Today, embolization is

an accepted treatment of anterior and posterior epistaxis,

where available. This report illustrates a case of epistaxis

caused by a radiation-induced and nontraumatic ICA

hemorrhage without presenting pseudoaneurysm.

2. Case report

An 80-year-old man was admitted in to our hospital with

a 1-week history of recurrent left-sided epistaxis. Eleven

years ago, the patient underwent a radical maxillectomy for

squamous cell carcinoma of the maxillary antrum. Postop-

erative radiotherapy had been given according to the

Available online at www.sciencedirect.com

American Journal of OtolaryngologyHead and Neck Medicine and Surgery 32 (2011) 162164www.elsevier.com/locate/amjoto

Corresponding author. Department of Otolaryngology-Head and Neck

Surgery, Eye, Ear, Nose and Throat Hospital, Shanghai Medical College,

Fudan University, 83 Fenyang Road, Shanghai 200031, PR China. Tel.: +86

21 64 377 134 388; fax: +86 21 64 377 151.

E-mail address: wangdehuient@sina.com (D.-H. Wang).

0196-0709/$ see front matter 2011 Elsevier Inc. All rights reserved.

doi:10.1016/j.amjoto.2009.10.006

mailto:wangdehuient@sina.comhttp://dx.doi.org/10.1016/j.amjoto.2009.10.006http://dx.doi.org/10.1016/j.amjoto.2009.10.006mailto:wangdehuient@sina.com

7/28/2019 14247 Non Trauma n Post Radiated Unusual Epis

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regulation dose. Two years ago, he received a course of

knife therapy for metastasis in the left temporal lobe of

the brain.

The initial physical examination showed unremarkable.

Contrast-enhanced computed tomography and magnetic

resonance imaging (MRI) scans revealed no hemorrhagic

focus. Interestingly, the MRI scan revealed that the lateral

wall of the sphenoid sinus was quite close to the ICA

(Fig. 1A). A review of his previous films revealed that only a

thin layer of soft tissue separated the ICA from the lateral

wall of the sphenoid sinus (Fig. 1B).

During the endoscopic operation, osteonecrosis was

found on the anterior skull base with a defect area of 2

3 cm2. Cerebral dura mater was exposed with obvious

pulsation. The operation ended by anterior and posterior

packing with yarn.

The patient was transferred to Shanghai Renji Hospital

Neurosurgery Department, where an emergent angiogram

revealed much leakage in the cavernous segment of the ICA.

After a successful balloon occlusion test with hypotensive

challenge, detachable balloon occlusion embolization of the

left ICA was performed. Two balloons were placed,

respectively, on the proximal and distal ends of that segment.The patient had no neurologic deficit as a result of the

procedure. A follow-up at 6 months demonstrated no

symptoms of epistaxis or neurologic deficit.

3. Discussion

Epistaxis, the most common emergency in otorhinolaryn-

gology [1], results from a multitude of causes, both local and

systemic. Intracavernous carotid hemorrhage is a rare cause

of epistaxis. The most common presentation for nontraumatic

and postirradiated cavernous ICA rupture is pseudoaneurysm

[6-8]. Our patient was a very unusual case of ICA hemorrhage

with leakage of the intracavernous carotid artery but no

pseudoaneurysm. Pseudoaneurysm formation results from

ICA hemorrhage or hematoma forming a peripheral fibrous

wall. Weakening and enlargement from continuous pulsatile

forces can result in breakdown of the fibrous wall with

eventual rupture. In cases of significant trauma to the anterior

cranial base, pseudoaneurysms can occur in the cavernous

segment of the ICA. The initial clinical presentation may be

massive epistaxis resulting from disruption through the

sphenoid sinus wall [3].

However, in this case with previous maxillectomy,

radiotherapy, and knife therapy, osteoradionecroses ofboth the maxilla and the skull base were found during

endoscopy. The base of the cavernous sinus had only a thin

layer of soft tissue separating the lateral wall of the sphenoid

sinus from the ICA. The hemorrhage from the ICA directly

entered the sphenoid sinus through the cavernous sinus,

making a cavernous internal carotid pseudoaneurysm

impossible.

Osteoradionecroses of the maxilla and base of skull are

rare phenomena, usually seen after combined therapies for

malignancies of the maxillary sinus. Although the mandible

is most commonly affected by osteoradionecroses, the

Fig. 1. Anteroposterior-view magnetic resonance imaging of ICA segment.

(A) A T2-weighted MRI reveals left nasal cavity was packed with yarn and

the lateral wall of the sphenoid sinus was close to the ICA (arrowhead). (B)

A T1-weighted MRI revealed only a thin layer of soft tissue between the

lateral wall of the sphenoid sinus and the ICA (arrowhead).

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maxilla and skull base may also be affected when

preoperative or postoperative radiotherapy is combined

with surgery. Contributing factors may include high

radiation dosage delivered to the treatment volume, loss of

tissue protective effects due to surgery, decreased vascularity

caused by surgery and radiation, and proximity of a

contaminated field. Onset of symptoms may vary and may

include pain, trismus, and purulent discharge. However, in

this case, only a headache was noted. The best diagnostic

modality remains the history and physical examination

because the area is readily accessible. Computed tomogra-

phy scans may help in diagnosis and treatment planning [9].

Lam et al [6] reported 4 cases of ICA pseudoaneurysm

rupture after radiation-induced temporal bone osteoradione-

crosis. They concluded that skull base osteoradionecrosis

with bleeding from the ICA is a potentially fatal complica-

tion of irradiation. Angiography was the mainstay of

diagnosis with embolization of the aneurysm and emboliza-

tion or ligation of the ICA as management options.

Embolization for epistaxis was first performed bySokoloff[10] in 1974. Since then, embolization has become

an accepted treatment of posterior epistaxis, where available

[11]. ICA occlusion is the definitive treatment in the ICA

hemorrhage, provided cross-circulation is adequate. Never-

theless, ligation or embolization occlusion of the ICA runs a

high risk of a cerebrovascular accident and mortality [6].

Acute ischemic infarcts causing death may occur once the

ICA is ligated or embolized. The likelihood of cerebral

complications depends on the adequacy of the collateral

blood supply. In this case, the successful balloon occlusion

test with hypotensive challenge minimized the likelihood of

inadequate cross-circulation. To our knowledge, old age hasnot been reported as a contraindication in performing ICA

embolization, and the procedure was successfully per-

formed in this 80-year-old man. In case of failure of this

procedure, ligation of ICA and extraintracranial bypass

surgery would have been considered, although the potential

risk and complication had to be fully discussed with the

patient and family.

Multidisciplinary collaboration is very important in

treating epistaxis. Epistaxis is usually first dealt with in the

ENT department, but ICA hemorrhage will necessitate

collaboration of an otorhinolaryngologist, neurosurgeon,

radiologist, anesthesiologist, nurses, and other team mem-

bers. Carotid angiography must be examined cautiously to

investigate for slight ICA leakage. ICA embolization is the

definitive treatment provided cross-circulation is adequate.

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