Case Report

Vocal Cord Paralysis as the First Sign of Spontaneous Carotid Dissection in a Patient With Extracranial Internal Carotid Artery Aneurysm

Vascular and Endovascular Surgery 2016, Vol. 50(1) 52-56 ª The Author(s) 2016 Reprints and permission: sagepub.com/journalsPermissions.nav DOI: 10.1177/1538574415627698 ves.sagepub.com

Petar Popov, MD, PhD1,2, Rene Chapot, MD3, Slobodan Tanaskovic´, MD1, Berislav Vekic´, MD, PhD4, Vuk Sotirovic, MD1, Nenad Ilijevski, MD, PhD1, and Djordje Radak, MD, PhD1,4

Abstract Introduction: Spontaneous dissection of supra-aortic arteries is an exceptionally rare cause of vocal cord dysfunction. We are reporting a case of spontaneous carotid dissection and internal carotid artery aneurysm presenting as vocal cord paralysis. Case Report: A 44-year-old female was admitted with hoarseness and swallowing disorders. Diagnostic imaging revealed dissection and obliteration of the right internal carotid artery (ICA) 23 mm from the carotid bifurcation. Electromyography revealed unilateral paralysis/paresis of the right vocal cord. Genetic analyses for thrombophilia, methylenetetrahydrofolate reductase, and plasminogen activator inhibitor 1 were found to be at high risk. The patient was discharged after 5 days without any neurological findings, and control angiography revealed complete restitution of the flow in the right ICA one month later. However, a fusiform aneurysm of the distal part of the extracranial right ICA was detected and excluded with endovascular procedure. Conclusion: Connective tissue systemic disorders and even mild trauma could initiate the dissection process of neck arteries. Precise diagnosis might be difficult even for an experienced neurologist, however, the final outcome is favorable. Keywords carotid artery, dissection, vocal cord, paralysis

Introduction Vocal cord paralysis as the first sign of spontaneous internal carotid artery dissection is extremely rare. Although it can occur at any age, dissection is most commonly seen in the fifth decade of life with an incidence of 2.6 to 2.9 patients per 100 0001-4 and men being more often affected than women (3:2).1 The disease starts with intimal tear or rapid splitting of the tunica media or subadventitial layer of the artery wall, creating a false lumen and/or hematoma leading to narrowing or thrombosis of the true lumen. Likewise, distal embolization and brain ischemia can occur. Dissection can start both in the extracranial and in the intracranial part of the supra-aortic arteries (10%).3-8 In the following hours and days, the most common clinical presentation is ipsilateral headache, neck and eye pain, ptosis, vision disturbances, dysfunction of the cranial nerves (IX-XII),6,8-12 and stroke.1,11,12 Quite often, pseudoaneurysm formation at the splitting site is seen.5,13 Dissection is most commonly caused by trauma or hereditary systemic diseases of the connective tissue. In such cases, antiplatelet therapy should be started immediately to prevent possible

ischemic attacks. While diagnosis can be difficult, but the final outcome and prognosis are usually benign.

Case Report A 44-year-old female was urgently admitted to the vascular surgery clinic after complaining of headache, occasional visual field defects in the form of glittering light and blurred vision, hoarseness, and swallowing disorders. The first symptoms 1

Vascular Surgery Clinic, ‘‘Dedinje’’ Cardiovascular Institute, Belgrade, Serbia School of Medicine, Belgrade University, Belgrade, Serbia 3 Klinik fu¨r Radiologie und Neuroradiologie, Alfried Crupp Krankenhouse Ru¨ttenschield, Essen, Deutchland 4 Surgery Department, DrDragisˇa Misˇovic´, University Medical Center, Baddour Parkway, LB, USA 2

Corresponding Author: Slobodan Tanaskovic´, Vascular surgery Clinic, Dedinje Cardiovascular Institute, Heroja Milana Tepica 1 Street, Belgrade 11000, Serbia. Email: [email protected]

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Figure 1. On the day of admission to the hospital (A) right internal carotid artery (ICA) occlusion, 23 mm cranially from the bulb (multidetector computer angiography [MDCT]); (B) ICA occlusion—petrous canal.

occurred 1 hour after an aerobic training. At admission, the patient was normotensive, without any apparent neurological disorders. Echocardiography was normal and laboratory analyses were within the expected values. She denied previous surgical interventions, history of neurological disorders, malignancies, trauma, family history, or smoking. Duplex ultrasonography of the neck arteries revealed suspected dissection and thrombosis in the proximal part of the extracranial right internal carotid artery (ICA). Multidetector computer angiography (MDCT) confirmed obliteration of the right ICA 23 mm from the carotid bifurcation (Figure 1). Neurological examination was normal. Brain computed tomography (CT) excluded intracranial bleeding or ischemia and therefore, anticoagulant therapy was administrated, Fondaparinux 5 mg once a day, overlapping with warfarin for 5 days. Because of hoarseness and minor swallowing problems, laryngoscopy and laryngeal electromyography were done, and unilateral paralysis/paresis of the right vocal cord was found. Genetic analyses for thrombophilia-inherited factor V Leiden and prothrombin G20210A mutation, methylenetetrahydrofolate reductase, and plasminogen activator inhibitor 1 were

found to be at high risk. Along with ICA aneurysm and mentioned dissection, this fact significantly contributed to ICA thrombosis. As the patient was asymptomatic, she was discharged after 5 days without any neurological findings. After 3 weeks, control duplex ultrasonography showed partial recanalization of the right ICA (2 of 3 of the lumen), with the false lumen being thrombosed. A month later, the MDCT angiography revealed complete restitution of the flow in the right ICA. However, a fusiform aneurysm of the distal part of the extracranial right ICA was detected, measuring 12.8  8.2 mm in diameter, without mural thrombus (Figure 2). Control brain CT showed regular findings. Aneurysm exclusion of the right ICA was obtained by placing 5  30 mm Cobalt-Chrome self-expandable Wallstent (Boston Scientific, Marlborough, Massachusetts, United States), with the previous application of 6 coils (hydrogelcoated coils; Microvention, Terumo, Tustin, California, United States) in the aneurysmal sac (Figure 3). Dual antiplatelet therapy (Aspirin and Clopidogrel) was administered for the next 6 months to prevent any possible post-procedural thrombosis.

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Figure 2. Multidetector computer angiography (MDCT) angiography after a month, pseudoaneurysm formation (12.8  8.2 mm) and complete internal carotid artery (ICA) recanalization (MDCT).

Hoarseness resolved completely, and the next otolaryngological examination showed complete restitution of the laryngeal muscle mobility and no right vocal cord paresis.

Discussion Only few cases of vocal cord paralysis/paresis resulting from of the ICA dissection are reported in the literature4,8-11 The most common causes of laryngeal dysfunction described are tumors of the neck, viral infections, neck surgery (including carotid reconstruction), trauma, and cerebral infarction. Dissections are often spontaneous and/or associated with ‘‘trivial trauma.’’12 In such a case, during casual body and neck movement or exercise, cervical arteries come in close contact with the transverse process of the second and third cervical vertebra or the styloid process.11,12,14 On the other hand, dissections can be seen in patients with some systemic diseases (Marfan syndrome, Ehlers Danlos syndrome IV, fibromuscular dysplasia, Behcet syndrome, and osteogenesis imperfecta type I) and connective tissue abnormalities12-15 as a result of genetic inheritance, the

connective tissue recomposition (abnormalities in types II and III, Ehlers Danlos Syndrome), and the reduced production of the type III collagen.12,13,15 In 15% to 25% of cases, fibromuscular dysplasia could be the cause of spontaneous carotid dissection.4 Inflammatory process in the tunica media and accompanying vasa vasorum were marked as the potential causes of arterial dissection, along with the aneurysmal sac development in patients with Behcet disease. Genetic abnormalities resulting in thrombophilia-inherited factor V Leiden and prothrombin G20210A mutation, as seen in our patient, might be the cause of carotid artery dissection and secondary thrombosis. Thrombophilia has already been described as one of the predisposing conditions to the development of arterial thrombosis16 which in addition to dissection and coexisting aneurysm in the presented case surely contributed to ICA thrombosis development. In the available literature we could not find a similar case to describe spontaneous dissection, vocal cord paralysis and extracranial ICA aneurysm with associated thrombophilia.12 Although uncommon, arterial dissection of cervical arteries should be considered in the differential diagnosis of a patient

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Figure 3. Seventy-five days later; cobalt-chrome self-expandable wallstent (5  30; Boston Scientific) with assisted coiling (6 coils; Microvention) involves permanent placement of a stent in the right internal carotid artery (ICA) pseudoaneurysm adjacent to the Pseudoaneurysm (PSA) to provide a scaffolding of support that keeps the coils within the aneurysm sac.

presenting with laryngeal dysfunction. In more than 20% of the cases, both carotid and vertebral artery dissection could be seen.11,13

Conclusion Cervical carotid artery dissection should be considered whenever vocal cord paralysis or paresis is diagnosed. Although descriptions of this entity are scarce in the literature, outcome appears favorable. Declaration of Conflicting Interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This manuscript was partly funded by Serbian Ministry of Science and technological developmentx—Project No 41002.

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7. Patel RR, Adam R, Maldjian C, Lincoln CM, Yuen A, Arneja A. Cervical carotid artery dissection: current review of diagnosis and treatment. Cardiol Rev. 2012;20(3):145-152. 8. Boukobza M, Ast G, Reizine D, Merland JJ. Internal carotid artery dissection causes hypoglossal nerve palsy: CT, MRI, and angiographic findings. J Neuroimaging. 1998;8(4):244-246. 9. Nakagawa H1, Kusuyama T, Ogawa K. Isolated vagus nerve paralysis associated with internal carotid artery dissection. Auris Nasus Larynx. 2014;41(1):118-120. 10. Peltz E, Ko¨hrmann M. Images in clinical medicine. Internalcarotid-artery dissection and cranial-nerve palsies. N Engl J Med. 2011;365(23):e43. 11. Caplan LR, Gonzalez RG, Buonanno FS. Case records of the Massachusetts General Hospital. Case 18-2012: A 35-year-old man with neck pain, hoarseness, and dysphagia. N Engl J Med. 2012;366(24):2306-2313.

12. Mayer SA, Rubin BS, Starman BJ, Byers PH. Spontaneous multivessel cervical artery dissection in a patient with a substitution of alanine for glycine (G13A) in the alpha 1 (I) chain of type I collagen. Neurology. 1996;47(2):552-556. 13. van den Berg JS, Limburg M, Kappelle LJ, Pals G, Arwert F, Westerveld A. The role of type III collagen in spontaneous cervical arterial dissections. Ann Neurol. 1998;43(4):494-498. 14. Wamuro Y, Nakahara I, Higashi T, et al. Spontaneous improvement of idiopathic intracranial carotid artery dissection: a case report. No Shinkei Geka. 2008;36(7):625-631. 15. Brandt T, Hausser I, Orberk E, et al. Ultrastructural connective tissue abnormalities in patients with spontaneous cervico cerebral artery dissections. Ann Neurol. 1998;44(2):281-285. 16. Previtali E, Bucciarelli P, Passamonti SM, Martinelli I. Risk factors for venous and arterial thrombosis. Blood Transfus. 2011; 9(2):120-138.

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Vocal Cord Paralysis as the First Sign of Spontaneous Carotid Dissection in a Patient With Extracranial Internal Carotid Artery Aneurysm.

Spontaneous dissection of supra-aortic arteries is an exceptionally rare cause of vocal cord dysfunction. We are reporting a case of spontaneous carot...
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