Primary Internal Carotid Artery Aneurysm in a 15-Year-Old Male: Case Report and Review of the Literature Daniel Lopez, Timur Sarac, and Robert Lorenz, Cleveland, Ohio

Extracranial internal carotid artery aneurysms are a rare entity in the adult population. Very little information is known in the pediatric population. We present a case of a 15-year-old male with an isolated internal carotid artery aneurysm and a review of the literature.

CASE REPORT A 15-year-old male presented to an outside facility in early 2013 for progressive swelling on the right side of his neck. The patient denied any neurologic complaints, and was right hand dominant. The patient denied any history of tonsillectomy, previous trauma, or any recent febrile illness. The patient was investigated with a computed tomography angiogram which revealed a 3.1  6.7-cm dysplastic aneurysm of the right internal carotid artery (Figs. 1 and 2) with return to normal caliber just below the skull base. The patient was placed on aspirin therapy at this time. The patient was referred to our clinic for a second opinion. Given the distal extent of the lesion, a multidisciplinary approach was pursued involving otolaryngology and vascular surgery. The operative plan was to perform direct reconstruction with reversed saphenous vein. Preoperatively, the patient had flexible laryngoscopy, which revealed normal cord function and normal superior laryngeal

Department of Vascular Surgery, Cleveland Clinic Foundation, Cleveland, OH. Correspondence to: Daniel Lopez, MD, Department of Vascular Surgery, Cleveland Clinic Foundation, 9500 Euclid Avenue, Desk F30, Cleveland, OH 44195, USA; E-mail: [email protected] Ann Vasc Surg 2015; 29: 126.e1e126.e4 http://dx.doi.org/10.1016/j.avsg.2014.07.039 Ó 2015 Elsevier Inc. All rights reserved. Manuscript received: June 1, 2014; manuscript accepted: July 29, 2014; published online: October 7, 2014 .

nerve function. Venous mapping revealed adequate vein for the planned procedure. The patient was taken to the operating room and a modified Blair incision was performed and the stylohyoid muscle and the posterior belly of digastric muscle were transected. To gain further access to the area, the styloid process was resected from the skull base exposing the full length of the aneurysm (Fig. 3). During the dissection, cranial nerves VII, IX, X, XI, and XII were identified and preserved; however, significant retraction of the nerves was required to gain access to the aneurysm. An Argyle shunt was inserted, and direct reconstruction was performed using reversed saphenous vein (Fig. 4). Postoperatively, the patient was placed in the pediatric intensive care unit. On physical examination, the patient had tongue deviation to the right, weakness in the right shoulder, difficulty swallowing, and clearing secretions. On postoperative day 1 the patient was taken back to the operating room for vocal cord injection with Radiesse voice gel to help augment his speech and help improve his cough. A nasojejunal tube was placed for nutritional support. The patient had a modified barium swallow study that showed normal swallowing function, and his diet was slowly advanced. The nasojejunal tube remained throughout his hospital course because of inadequate caloric intake. The patient was discharged on postoperative day 7, and aspirin therapy was continued. The patient was reevaluated on an outpatient basis on postoperative day 15. Swallow study revealed no evidence of aspiration and the nasojejunal tube was removed. The patient 126.e1

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Fig. 1. Coronal view of right internal carotid artery aneurysm.

did have some improvement in his cranial nerve function but had residual tongue deviation and weakness in the trapezius muscle. The patient was planned to have follow-up carotid duplex 3 months postoperatively but has been lost to follow-up.

DISCUSSION The first successful surgery for extracranial carotid artery aneurysms (ECAAs) was performed in 1808 by Sir Astley Cooper where the internal carotid artery was ligated successfully.1 Subsequent to this, Winslow performed a review of ECAAs demonstrating a 71% mortality rate with observation alone.2 In a more modern series, Zwolak reported a 50% stroke rate with observation alone.3 Ligation has also been reported in more modern series but it carries a high mortality rate.4e6 Although there have been some case reports in the pediatric literature, which suggest that ligation may be a viable option with little neurologic consequence,7e9 its use must be tempered against the patient’s surgical risk, accessibility of the lesion, and status of the Circle of Willis. It was not until the 1970s that direct reconstruction or autogenous vein grafting had replaced ligation for the management of these lesions.10 Surgical treatment for ECAAs in adolescence is rare because of the very low incidence and relative paucity of literature on the subject. Most data dictating the repair and management of

Annals of Vascular Surgery

Fig. 2. Axial view of right internal carotid artery aneurysm.

these lesions come from the adult literature with scattered case reports of repair in children.7e9,11 There is a considerable overlap between symptoms and postoperative results between these 2 populations. However, there are some noteworthy differences between the 2 groups. The accepted definition of what constitutes an ECAA comes from DeJong et al.12 stating that aneurysms in the region of the bulb have a diameter of more than 150% of the normal common carotid artery or more than 200% of the normal internal carotid artery. For the internal carotid artery, dilatations of more than 120% of a normal cross section of the ipsilateral artery are considered aneurysmal. This is a departure from the traditional definition of other peripheral artery aneurysms. In the adult population, most aneurysms are false aneurysms after carotid endarterectomy. In the largest series, false aneurysms comprised 56% of the cases.4 However, in more modern series, atherosclerotic lesions and dysplastic lesion comprise the majority.13,14 In the pediatric population, the major causes are congenital, traumatic, or mycotic in nature. There is a male predominance of these lesions of almost 2:1.15 The most common symptom in both the adult and the pediatric population is a pulsatile neck mass. Cranial nerve compression rates range between 4.6% and 11%.4,16 Although rupture is a relatively rare presentation for an adult,

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Fig. 3. Operative exposure of the internal carotid artery aneurysm.

Fig. 4. Final reconstruction with reversed saphenous vein graft.

Pourhassan’s review highlights the different nature of these lesions demonstrating that up to 42% of children present with complications from rupture of the aneurysm.11 Challenges of the open surgical approach include: injury to the cranial nerves, early neurologic complications due to intraoperative emboli, and exposure. Most modern series report an incidence of cranial nerve injury from 4% to 22% and a central neurologic complication rate ranging from 4% to 11%.13,14,16e19 Doing the distal anastomosis form inside the sac, as advocated by Moreau et al,13 decreases the risk of cranial nerve damage inherent to the isolation and control of the distal neck of the aneurysm. Compared with the rates of neurologic complications with observation alone it is clear that these lesions should be repaired at the time of their discovery. The low risk profile of the

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procedure and the high primary patency rates of the reconstructed segment20 favor repair of these lesions. Exposure of these lesions can be achieved through a standard carotid artery exposure, but as demonstrated in our case, consultation with otolaryngology can be essential in the exposure of lesions that extend distally or up to the skull base. Endovascular repair has been attempted to mitigate the complications of open surgery. May et al.21 reported the use of a stent graft for the exclusion of an ECAA. Unfortunately, the patient suffered distal embolization and subsequent thrombosis of the stent graft 6 months postoperatively. Subsequent case series22e24 have demonstrated decreased postoperative complications namely embolic phenomena with and without the use of neuroprotection devices. Zhou et al.25 compared the rates of postoperative complications with both open surgery and endovascular approaches and found that cranial nerve injuries decreased to 5% compared with 14% with open repair in their series. The use of endovascular techniques in children is less well defined. Wolfe et al.26 have reported the use of a stent graft for the treatment of a 10-year-old male with a large calcified internal carotid artery aneurysm with good results at 6 months. Furthermore, Gralla et al.27 reported the use of a stent graft in the treatment of a high internal carotid pseudoaneurysm in an 11-year-old with good results after 2 years. Although endovascular approaches are feasible and are an attractive alternative to open repair, there are questions that remain that will require further investigation for their use in the pediatric population. Namely, the natural history of the stent graft as the child grows, timing, and duration of antiplatelet therapy. A protocol for surveillance is another area in question that will need to be investigated. The use of a stent graft may be considered in the pediatric patient who is approaching adulthood, thus minimizing the amount of growth differential between the stent and the artery and high-risk surgical patients. REFERENCES 1. Cooper A. Account of the first successful operation performed on the common carotid artery for aneurysm in the year 1808. Guys Hosp Rep 1836;1:53e9. 2. Winslow H. Extracranial aneurysm of the internal carotid artery: history and analysis of the cases registered up to Aug 1, 1925. Arch Surg 1926;13:689e729. 3. Zwolak R, Whitehouse W, Knake J, et al. Atherosclerotic extracranial carotid artery aneurysms. J Vasc Surg 1984;1: 415e22. 4. El-Sabrout R, Cooley DA. Extracranial carotid artery aneurysms: Texas Heart Institute experience. J Vasc Surg 2000;31:702e12.

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5. Miksic K, Flis V, Kosir G, et al. Surgical aspects of fusiform and saccular extracranial carotid artery aneurysms. Cardiovasc Surg 1997;5:190e5. 6. Davidovic L, Kostic D, Maksimovic Z, et al. Carotid Artery Aneurysms. Vascular 2004;12:166e70. 7. Hazarika P, Sahota J, Nayak D, et al. Congenital internal carotid artery aneurysm. Int J Pediatr Otorhinolaryngol 1995;109:63e8. 8. Antar K, Keiser H, Peeva E. Relapsing arterial aneurysms in juvenile Bechet’s disease. Clin Rheumatol 2005;114:72e5. 9. Tovi F, Leiberman A, Hertzanu Y, et al. Pseudoaneurysm of the internal carotid artery secondary to tonsillectomy. Int J Pediatr Otorhinolaryngol 1987;13:69e75. 10. Hertzer N. Extracranial carotid aneurysms: a new look at an old problem. J Vasc Surg 2000;31:823e5. 11. Pourhassan S, Grotemeyer D, Fokou M, et al. Extracranial carotid arteries aneurysms in children: single-center experiences in 4 patients and review of the literature. J Pediatr Surg 2007;42:1961e8. 12. De Jong KP, Zondervan P, Van Urk H. Extracranial carotid artery aneurysms. Eur J Vasc Surg 1989;3:557e62. 13. Moreau P, Albat B, Thevenet A. Surgical treatment of extracranial internal carotid artery aneurysm. Ann Vasc Surg 1994;8:404e16. 14. Rosset E, Albertini J, Magnan E, et al. Surgical treatment of extracranial internal carotid artery aneurysms. J Vasc Surg 2000;31:713e23. 15. McCann RL. Basic data related to peripheral artery aneurysms. Ann Vasc Surg 1990;5:190e5. 16. Rhodes E, Stnaley J, Hoffman G, et al. Aneurysms of extracranial carotid arteries. Arch Surg 1976;111:339e43. 17. McCollum C, Wheeler W, Noon G, et al. Aneurysms of the extracranial carotid artery. Twenty-one years’ experience. Am J Surg 1979;137:196e200.

Annals of Vascular Surgery

18. Knight G, Hallman G, Reul G, et al. Surgical management of extracranial carotid artery aneurysms: report of 17 cases. Tex Heart Inst J 1988;15:91e7. 19. Faggioli G, Freyrie A, Stella A, et al. Extracranial internal carotid artery aneurysms: results of a surgical series with long term follow-up. J Vasc Surg 1996;23: 587e95. 20. Srivastava S, Eagleton M, O’Hara P, et al. Surgical repair of carotid artery aneurysms: a 10 year, single-center experience. Ann Vasc Surg 2010;24:100e5. 21. May J, White G, Waugh R, et al. Endoluminal repair of internal carotid artery aneurysm: a feasible but hazardous procedure. J Vasc Surg 1997;26:1055e9. 22. Bergeron P, Khanoyan P, Meunier J, et al. Long-term results of endovascular exclusion of extracranial internal carotid artery aneurysms and dissecting aneurysms. J Interv Cardiol 2004;17:245e52. 23. Juszkat R, Wrobel M, Golusinski W, et al. Stent-graft treatment of extracranial internal carotid artery aneurysm. Eur Arch Otorhinolaryngol 2005;262:826e9. 24. Szopinski P, Ciostek P, Kielar M, et al. A series of 15 patients with extracranial carotid artery aneurysms: surgical and endovascular treatment. Eur J Vasc Endovasc Surg 2005;29:256e61. 25. Zhou W, Lin P, Bush R, et al. Carotid artery aneurysm: evolution of management over two decades. J Vasc Surg 2006;43:493e6. 26. Wolfe S, Meuller-Kronast N, Aziz-Sultan A, et al. Extracranial carotid artery pseudoaneurysm presenting with embolic stroke in a pediatric patient. J Neurosurg Pediatr 2008;1: 240e3. 27. Gralla J, Brekenfeld C, Schmidli J, et al. Internal carotid artery aneurysm with life threatening hemorrhages in a pediatric patient. J Endovasc Surg 2004;11:734e8.

Primary internal carotid artery aneurysm in a 15-year-old male: case report and review of the literature.

Extracranial internal carotid artery aneurysms are a rare entity in the adult population. Very little information is known in the pediatric population...
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