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American Journal of OtolaryngologyHead and Neck Medicine and Surgery 32 (2011) 162 164 www.elsevier.com/locate/amjoto

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,
a

Department of Otolaryngology-Head and Neck Surgery, Eye, Ear, Nose and Throat Hospital, Shanghai Medical College, Fudan University, Shanghai, PR China b Department of Neurosurgery, Jiao Tong University, Shanghai, PR China c Division 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 80year-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 otorhinolaryngology [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 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

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. Postoperative radiotherapy had been given according to the

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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 otorhinolaryngology [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 of both 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 tomography 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 osteoradionecrosis. They concluded that skull base osteoradionecrosis with bleeding from the ICA is a potentially fatal complication of irradiation. Angiography was the mainstay of diagnosis with embolization of the aneurysm and embolization or ligation of the ICA as management options. Embolization for epistaxis was first performed by Sokoloff [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. Nevertheless, 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 has not been reported as a contraindication in performing ICA embolization, and the procedure was successfully performed 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 members. Carotid angiography must be examined cautiously to investigate for slight ICA leakage. ICA embolization is the definitive treatment provided cross-circulation is adequate. References
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