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Objective: To describe a rare case of refractory epistaxis due to a

pseudoaneurysm of the petrous ICA following radiation therapy.

Study Design: Case report and review of literature.

Method: Review of literature on Pubmed from 1990—present.

Case: A 54-year-old woman presented with copious right-sided epistaxis of 5-hours duration. She had sought prior treatment for recurrent unilateral epistaxis multiple times over a 3-month period and had undergone right sphenopalatine artery ligation. Her past medical history was notable for nasopharyngeal carcinoma with radiation therapy 11-years prior. MRI did not demonstrate tumor recurrence. Angiography of the right internal maxillary artery distribution revealed a pseudoaneurysm of the petrous ICA. Sequential coil-embolization of the pseudoaneurysm resolved the epistaxis.

Results: ICA pseudoaneurysm is a rare sequela following traumatic facial injury, sinonasal surgery, deep neck space infection, and even more rarely following radiation therapy. A total of 116 cases of internal carotid artery pseudoaneurysm were identified in the literature review. These cases were associated with trauma (n= 68), infection (n=18), iatrogenic (n=16), spontaneous dissection (n=8), radiation therapy (n=3), tumor invasion (n=2), pregnancy (n=1).

Conclusion: ICA pseudoaneurysm is an uncommon complication of XRT in patients with nasopharyngeal carcinoma. Optimal management demands rapid recognition, but prompt diagnosis of cavernous ICA pseudoaneurysm is often a clinical challenge. Because this problem is related to skull-base osteoradionecrosis, it may present as a long-term complication of radiation therapy. Otolaryngologists should be aware of this possible etiology in patients with refractory epistaxis and a history of previous radiation.

Refractory epistaxis resulting from internal carotid artery (ICA) pseudoaneurysm: A case report and review of literature.

Soroush Zaghi, MD1; Jonathan Yousefzadeh1; Bob Armin, MD1; Marilene B.Wang, MD1

1Department of Head and Neck Surgery, David Geffen School of Medicine at UCLA, Los Angeles, CA

Traumatic injury is the most commonly associated factor associated with ICA pseudoaneurysm. Massive epistaxis following blunt craniofacial trauma should alert clinicians to the possibility of traumatic (ICA) pseudoaneurysm2.

Deep neck space infection8, retropharyngeal abscess,9peritonsillar abscess, invasive fungal sinusitis10 and chronic otitis media11 have all been associated with life-threatening ICA pseudoaneurysm. Patients who present with ICA pseudoaneurysm associated with an infectious source have a notably high mortality rate with often grave complications when attempting endovascular approaches for treatment 12, 13.

Cases of ICA pseudoaneurysm following malignant tumor invasion have also been reported: recurrent oropharyngeal cancer encasing internal and external carotid artery14; sarcomatous transformation of prolactinoma15. However, authors note that the progression of the pseudoaneurysm may also have been due to surgical manipulation or radiotherapy exposure.

There are three prior case reports of ICA pseudoaneurysms following radiotherapy for the treatment of nasopharyngeal carcinoma 1 16 17; all three patients presented with refractory epistaxis. Interval from completion of radiotherapy ranged from three months to ten years. Iatrogenic factors besides radiotherapy have also been reported to cause ICA pseudoaneurysms. Arterial injury causing pseudoaneurysm is a well-known risk of angiography and endovascular therapy. In addition, biopsies of the oropharynx16, mandibular titanium plate reconstruction19, 20 , transsphenoidal surgery21, tonsillectomy 22, tympanic membrane myringotomy23, and internal jugular vein cannulation24 25

have all been associated with ICA pseudoaneurysm.

Irwin and Jacocks (2000) describe a case of ICA pseudoaneurysm during the pregnancy of a healthy 35 year-old at 40 weeks gestation. It was suggested that hemodynamic, hormonal, or other physiologic changes of pregnancy may weaken the arterial wall with increasing gestational age making pregnant women more susceptible to aneurysms 26.

An aneurysm is an abnormal widening or ballooning of a portion of an artery due to weakness in the wall of the blood vessel. A true aneurysm is one that involves all three layers of the wall of an artery (media, intima, adventitia). A pseudoaneurysm, or false aneurysm, occurs as the result of a leaking hole in a blood vessel. A hematoma forms outside the arterial wall such that leaking blood is contained by the surrounding tissues while remaining in continuity with the breached arteriallumen. This collection of blood may clot enough to seal the leak or may rupture the tougher tissue enclosing it and flow freely between layers of other tissues.

Pseudoaneurysm of the internal carotid artery is a rare sequela following traumatic facial injury, sinonasal surgery, deep neck space infection, and even more rarely following radiation therapy. Skull base osteoradionecrosis is a possible long-term complication after radiotherapy for nasopharyngeal carcinoma (NPC) that may contribute to this presenation18 ; the disorder is characterized by bony destruction or sequestration associated with the skull base and common symptoms include foul odor, headache, and epistaxis 18. Angiography and endovascular embolization is an effective diagnostic and treatment modality for the treatment of ICA pseudoaneurysm, but also carries significant risks. Surgical treatment proves to be difficult due to the fibrous tissue surrounding the carotid artery as well as the fragile nature of the irradiated carotid artery wall 17.

More than 60% of the adult population experience at least one episode of epistaxis during their lifetime 1. Most cases of epistaxis occur in the anterior septal area at Kiesselbach's plexus, or Little’s Area, which is a localized region of mucosa of the anteroinferior nasal septum supplied by branches of the sphenopalatine, greater palatine, and facial arteries.

Epistaxis in this location is readily accessible and treatable by cautery or anterior nasal packing. Posterior epistaxis often requires more aggressive measures including posterior nasal packing and endoscopic cauterization. Epistaxis refractory to initial treatment attempts can often be successfully treated by endovascular embolization techniques.

Internal carotid artery pseudoaneurysm is a rare and potentially fatal cause of epistaxis that demands prompt recognition. Although a majority of ICA pseudoaneurysms are associated with trauma or infection, cases associated with pregnancy, iatrogenic injury, tumor invasion, and radiotherapy have also been reported.

Here we report a case of refractory epistaxis in a patient with a history of radiation therapy so as to raise awareness of this potential etiology of epistaxis among patients treated for head and neck cancer.

INTRODUCTION

1. Buyukcam F, Sonmez FT, Aydin K. Successfully treated massive epistaxis in a patient with internal carotid artery pseudoaneurysm. J Craniofac Surg. Jul 2010;21(4):1304-1305.

2. Tseng YY, Yang ST, Yeh YS, Yang TC, Wong HF. Traumatic internal carotid artery pseudoaneurysm mimicking sphenoid sinus tumor. Rhinology. Dec 2007;45(4):332-334.

3. Thomas JA, Ware TM, Counselman FL. Internal carotid artery pseudoaneurysm masquerading as a peritonsillar abscess. J Emerg Med. Apr 2002;22(3):257-261.

4. Ahmed B, Perry M, Shetty S. Interesting Case: Pseudoaneurysm of internal carotid artery after severe maxillofacial injury which caused superior orbital fissure syndrome. Br J Oral MaxillofacSurg. Aug 2006;44(4):316.

5. Singh RR, Thomas AA, Barry MC, Bouchier-Hayes DJ. Traumatic pseudoaneurysm of the internal carotid artery presenting with oculosympathetic palsy. Ir J Med Sci. Jul-Sep 2004;173(3):162-163.

6. Nusynowitz RN, Stricof DD. Pseudoaneurysm of the cervical internal carotid artery withassociated hypoglossal nerve paralysis. Demonstration by CT and angiography. Neuroradiology. 1990;32(3):229-231.

7. Gupta V, Niranjan K, Rawat L, Gupta AK. Stent-graft repair of a large cervical internal carotid artery pseudoaneurysm causing dysphagia. Cardiovasc Intervent Radiol. May 2009;32(3):558-562.

8. da Silva PS, Waisberg DR. Internal carotid artery pseudoaneurysm with life-threatening epistaxis as a complication of deep neck space infection. Pediatr Emerg Care. May 2011;27(5):422-424.

9. Beningfield A, Nehus E, Chen AY, Yellin S. Pseudoaneurysm of the internal carotid artery after retropharyngeal abscess. Otolaryngol Head Neck Surg. Feb 2006;134(2):338-339.

10. Jao SY, Weng HH, Wong HF, Wang WH, Tsai YH. Successful endovascular treatment of intractable epistaxis due to ruptured internal carotid artery pseudoaneurysm secondary to invasive fungal sinusitis. Head Neck. Mar 2011;33(3):437-440.

11. Oyama H, Hattori K, Tanahashi S, Kito A, Maki H, Tanahashi K. Ruptured pseudoaneurysm of the petrous internal carotid artery caused by chronic otitis media. Neurol Med Chir (Tokyo). 2010;50(7):578-580.

12. Biron A, Berkowitz RG, Bekhit EK, Rose EA. Ultrasound diagnosis of an internal carotid arterypseudoaneurysm in a young child. Int J Pediatr Otorhinolaryngol. Nov 2006;70(11):1975-1979.

13. Semple CW, Berkowitz RG, Mitchell PJ. Embolization of an extracranial internal carotid artery pseudoaneurysm. Ann Otol Rhinol Laryngol. Feb 2005;114(2):90-94.

14. Kakizawa H, Toyota N, Hieda M, et al. Massive hemorrhage from internal carotid artery pseudoaneurysm successfully treated by transcatheter arterial embolization with assessment of regional cerebral oxygenation. Cardiovasc Intervent Radiol. Jul-Aug 2005;28(4):495-498.

15. Tachibana E, Saito K, Wakabayashi T, Nagasaka T, Furui T, Yoshida J. Sarcomatous transformation of a prolactinoma associated with development of a fatal internal carotid artery pseudoaneurysm--case report. Neurol Med Chir (Tokyo). Aug 2000;40(8):427-431.

16. 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. Aug 2008;30(8):1132-1135.

17. Kiyosue H, Okahara M, Tanoue S, et al. Dispersion of coils after parent-artery occlusion of radiation-induced internal carotid artery pseudoaneurysm. AJNR Am J Neuroradiol. Jun-Jul 2004;25(6):1080-1082.

18. Huang XM, Zheng YQ, Zhang XM, et al. Diagnosis and management of skull base osteoradionecrosis after radiotherapy for nasopharyngeal carcinoma. Laryngoscope. Sep 2006;116(9):1626-1631.

19. Cagici CA, Kizilkilic O, Yavuz H, Oguzkurt L, Giray S, Ozluoglu L. [Pseudoaneurysm of the internal carotid artery following a tonsil biopsy: endovascular repair with covered stent]. Kulak Burun Bogaz Ihtis Derg. Mar-Apr 2008;18(2):101-105.

20. DeFatta RJ, Verret DJ, Bauer P. Extracranial internal carotid artery pseudoaneurysm. Int J Pediatr Otorhinolaryngol. Aug 2005;69(8):1135-1139.

21. Hattori I, Iwasaki K, Horikawa F, Tanji M, Gomi M. [Treatment of a ruptured giant internal carotid artery pseudoaneurysm following transsphenoidal surgery: case report and literature review]. No Shinkei Geka. Nov 2006;34(11):1141-1146.

22. Raffin CN, Montovani JC, Neto JM, Campos CM, Piske RL. Internal carotid artery pseudoaneurysm after tonsillectomy treated by endovascular approach. A case report. IntervNeuroradiol. Mar 30 2002;8(1):71-75.

23. Henriksen SD, Kindt MW, Pedersen CB, Nepper-Rasmussen HJ. Pseudoaneurysm of a lateral internal carotid artery in the middle ear. Int J Pediatr Otorhinolaryngol. Apr 15 2000;52(2):163-167.

24. Nayeem SA, Tada Y, Takagi A, Sato O, Miyata T, Idezuki Y. Carotid artery pseudoaneurysm following internal jugular vein cannulation. J Cardiovasc Surg (Torino). Mar-Apr 1990;31(2):182-183.

25. Mastan M. Carotid artery pseudoaneurysm with Horner's syndrome: delayed complication of internal jugular venous cannulation. Hosp Med. May 2005;66(5):314-315.

26. Irwin RJ, Jacocks MA. Internal carotid artery pseudoaneurysm related to pregnancy. Ann VascSurg. Jul 2000;14(4):405-409.

27. Koroglu M, Arat A, Cekirge S, et al. Giant cervical internal carotid artery pseudoaneurysm in a child: endovascular treatment. Neuroradiology. Oct 2002;44(10):864-867.

28. Tseng A, Ramaiah V, Rodriguez-Lopez JA, et al. Emergent endovascular treatment of a spontaneous internal carotid artery dissection with pseudoaneurysm. J Endovasc Ther. Jun 2003;10(3):643-646.

DISCUSSION

LITERATURE REVIEW

REFERENCES

Figure 3. Cerebral Angiography Image 3: Treatment involves placement of platinum coils to achieve hemostasis and thrombus formation. A) The pseudoaneurysm was catheterized and GDC platinum coils were deployed within the hematoma cavity. B) Following sequential coil embolization, interval angiograms demonstrate progressive thrombosis within the aneurysm dome. Definitive therapy involves placement of a stent across this portion of the ICA. Intravascular stent placement is delayed for 4-6 weeks until after the resolution of the acute bleed. Definitive stent placement requires the initiation of antiplatelet therapy, which is contraindicated in the acute setting.

Figure 1. Cerebral Angiography Image 1: A microcatheter is threaded through the femoral artery and radiologically guided to the right common carotid artery. Contrast is ejected to evaluate vascular blushing of the right internal maxillary artery distribution. Images from the lateral transnasal angiogram are displayed: A) Two overlapping rhinorockets are seen in the right nasal cavity. B) Otherwise, normal vasculature of the nasal mucosa.

ABSTRACT

Soroush Zaghi, MD.Resident PhysicianUCLA Dept. of Head and Neck SurgerySzaghi@mednet.ucla.edu

CONTACT

1. A. 1. B.

2. A. 2. B.

3. A. 3.B.

Figure 2. Cerebral Angiography Image 2: Images from the AP intracranial angiogram of the right common carotid artery are displayed: A) There is an unusually shaped area of contrast stagnation at the petrous segment of the right internal carotid. B) This portion of the artery appears to fill slowly and retains contrast for longer duration. The images are consistent with a 5 mm x 9mmpseudoaneurysm which arises from the medial wall of the verticalsegment of the petrous ICA. There are no intimal flaps or intraluminal filling defects consistent with either thrombus or dissection within the parent artery.