The Laryngoscope C 2014 The American Laryngological, V
Rhinological and Otological Society, Inc.
Cervical Internal Carotid Artery Pseudoaneurysm Complicating Malignant Otitis Externa: First Case Report Andrew Baker, BS; Habib Rizk, MD; William Carroll, MD; Paul Lambert, MD Pseudoaneurysm of the internal carotid artery (ICA) is a rare complication of head and neck infections. To date, three cases of petrous ICA pseudoaneurysm have been described as a complication of otogenic infection, including only one secondary to malignant otitis externa. We present here the first case of cervical ICA pseudoaneurysm as a complication of malignant otitis externa, and stress the importance of timely diagnosis to avoid fatal outcomes. Key Words: Internal carotid artery, pseudoaneurysm, malignant otitis externa. Laryngoscope, 125:733–735, 2015
INTRODUCTION Pseudoaneurysm of the internal carotid artery (ICA), first described by Liston, is a rare complication of head and neck infections.1 Between 1950 and 2012 there were 99 reported cases of extracranial carotid artery pseudoaneurysm. These pseudoaneurysms can arise from septic emboli, trauma, or iatrogenic causes; however, most are caused by direct spread of local infections.2 Currently, pseudoaneurysms most commonly arise from local infections in children.3 To date, three cases of petrous ICA pseudoaneurysm have been described as a complication of otogenic infection including one following chronic otitis media, one consequent of tuberculous otitis, and one secondary to malignant otitis externa (MOE).4–6 One additional case of extracranial ICA pseudoaneurysm at the level of the jugular foramen has been described following an acute otitis media.7 The case presented here illustrates the rare event of pseudoaneurysm of the cervical ICA as a complication of malignant otitis externa from Pseudomonas aeruginosa.
CASE REPORT An 81-year-old male with a history of bilateral, moderate to profound, sensorineural hearing loss and non–insulin-dependent diabetes mellitus type 2 initially presented to an outside otolaryngologist with left otalgia.
From the Department of Otolaryngology–Head and Neck Surgery, Medical University of South Carolina, Charleston, South Carolina, U.S.A The authors have no funding, financial relationships, or conflicts of interest to disclose. Editor’s Note: This Manuscript was accepted for publication July 21, 2014. Send correspondence to Andrew Baker, Otolaryngology–Head and Neck Surgery, Medical University of South Carolina, 135 Rutledge Avenue, MSC 550, Charleston, SC 29425. E-mail: [email protected]
Laryngoscope 125: March 2015
He was treated topically for otitis externa and underwent tympanostomy tube placement for a left middle ear effusion. Computed tomography (CT) imaging showed mastoid opacification without any bony erosion. When he presented to the otology division at our tertiary care center 2 months later, he complained of persistent left otalgia, left sided tinnitus, and trismus. On exam he was found to have purulent drainage in his left external auditory canal (EAC), with a large polyp in the medial third. There was no granulation tissue present. His outside CT and magnetic resonance imaging (MRI) were both reviewed and demonstrated inflammation within the left mastoid cavity and temporomandibular joint capsule edema, with no bony erosions noted and no venous sinus thrombosis. Two wicks were placed in his ear, and he was started on antibiotic/steroid drops and oral fluoroquinolone due to suspicion for malignant otitis externa. He had a brief inpatient stay shortly thereafter for presumed reaction to the fluoroquinolone. His hemoglobin A1c at that time was 6.4. He was switched to cefdinir and discharged. When seen 1 week later, his polyp had resolved and his pain was significantly improved. Mild granulation tissue was noted and he was started on acetic acid irrigations and continued Ciprodex drops. Cultures from his EAC were consistently negative. On his subsequent follow-up visit (3 weeks after his initial presentation to our institution), he had recurrence of polypoid tissue in the left EAC that was biopsied. Pathology showed granulation tissue with acute inflammation. A CT scan was ordered at that time, but he was admitted to an outside hospital with altered mental status and sepsis before the scan was obtained. An MRI scan was obtained acutely and showed a new left parapharyngeal mass and two small acute left strokes: one in the parietal cortex and one adjacent to the left lateral ventricle. Blood cultures were obtained and were positive for P aeruginosa sensitive to levofloxacin, imipenem, cefepime, and piperacillintazocin; he was started on cefepime. Baker et al.: ICA Pseudoaneurysm in MOE
Fig. 1. (A, B) Pre-embolization magnetic resonance imaging (MRI) of the patient shows a large left internal carotid artery pseudoaneurysm. (C, D) Post-embolization MRI shows a thrombosed pseudoaneurysm and stable soft tissue filling the left middle ear cavity, eustachian tube, and nasopharynx. (A, C) Axial T2. (B, D) Axial T1 with contrast.
He was eventually transferred to our tertiary care center. On presentation, he had a mild alteration of mental status. His soft palate deviated to the right. Flexible nasopharyngolaryngoscopy showed effacement of the left nasopharynx with overlying blood streaks as well as an immobile left true vocal cord. Review of his outside imaging was consistent with a pseudoaneurysm of the distal cervical segment of the left ICA measuring 2.6 3 3.1 cm. Neurointerventional radiology consult was obtained, and he underwent coil embolization at the midcervical segment of the left ICA after a successful balloon occlusion test (Fig. 1). He was continued on cefepime, and otoscopy showed no signs of inflammation or polyp. A percutaneous endoscopic gastrostomy tube was placed 4 days into hospitalization because of his swallowing problems and his immobile vocal cord. On hospital day 25 he was discharged to a rehabilitation facility still on intravenous cefepime through a peripherally inserted central catheter (PICC) line and ciprodex drops in his left ear. His antibiotic treatment was subseLaryngoscope 125: March 2015
quently changed to meropenem. Follow-up MRI done 6 weeks after discharge showed slight improvement of local inflammation with stable coiled aneurysm. Clinically, the patient had a slow recovery. On evaluation 7 weeks after discharge (approximately 6 months after onset of symptoms), he was alert and oriented with no sequelae of his ischemic cerebrovascular accidents. He had no otalgia, trismus, or other ear complaints. His external auditory canal and tympanic membrane showed no evidence of inflammation. The PICC line was removed and he was given a 2month course of oral ciprofloxacin pending a follow-up MRI scan. The pseudoaneurysm complication did not alter the duration of antibiotic management of MOE.
DISCUSSION In a review of 22 patients, Guevara reported a mean time to diagnosis of MOE of 13 weeks.8 Early diagnosis is critical for eradication of the infection and to prevent complications. Cases of otitis externa refractory to initial Baker et al.: ICA Pseudoaneurysm in MOE
treatment should, therefore, be investigated by culture, CT scan, and/or MRI to rule out MOE.9 Cranial nerve palsy in the elderly, diabetic, or immunocompromised patient with imaging findings of bony destruction should prompt a workup for skull base osteomyelitis/MOE and malignancy. MOE should be treated with a culture-directed long-term antibiotic regimen and frequent debridement as necessary. P aeruginosa is the most common pathogen in MOE9; although, a recent report indicates increasing incidence of nonpseudomonal species in MOE, including methicillinresistant Staphylococcus aureus.10 MOE is a devastating disease with high mortality potential. In a 26-patient series, mortality of MOE was 19% with skull base osteomyelitis, intracranial extension, and cranial nerve involvement correlating with worse outcomes.11 In a cohort of 57 patients, 5-year survival was 55% without persistent disease and 40% with persistent disease (P 5.086). Risk of mortality was significantly increased in the >70-year-old age group and in those with persistent disease.12 MOE in our case was complicated by development of a pseudoaneurysm of the cervical ICA with an ischemic cerebrovascular event, approximately 3 months after initial presentation of otitis externa. This is the first reported case of cervical ICA pseudoaneurysm in the setting of a malignant otitis externa. There is one previous reported case of a petrous ICA pseudoaneurysm following a malignant otitis externa and skull base osteomyelitis.4 Pseudoaneurysms of the cervical internal carotid artery have been previously reported following deep neck space infections. They affect mostly children and result from a parapharyngeal abscess or an adjacent suppurative cervical adenitis.3 Cervical ICA pseudoaneurysms typically present as a pulsatile cervical mass. They may be associated with pain, tenderness, fever, dysphagia, dysphonia, recurrent hemorrhage, anemia, lower cranial nerve palsy, Horner syndrome, and incomplete resolution following full antibiotic course.2 In our case, the patient presented with otalgia, tinnitus, trismus, and eventually a parapharyngeal mass with 10th cranial nerve palsy. Notably, our patient did not endorse neck discomfort, one of the classic features of an ICA pseudoaneurysm. In fact, the only indication for CT angiography in this patient was the stroke. Although CT angiography is the preferred method for the diagnosis of a pseudoaneurysm, a CT or MRI scan should be able to demonstrate a neck mass suspicious for a pseudoaneurysm.13 Endovascular occlusion of an ICA pseudoaneurysm has become a common treatment. Currently, numerous cases of successful management with coiling have been reported.3–5,7 Endovascular management with a covered stent has also been shown to successfully treat ICA pseudoaneurysm while preserving blood flow through the affected artery.14 However, it is discouraged in an infectious context. Had this patient failed the balloon occlusion test, given that he is a poor surgical candidate, a covered stent would have been the next best option. Duration of MOE treatment is often difficult to follow as changes occur more slowly than improvement in clinical measures. CT is more sensitive to bone erosion and decreased bone density and is thus more useful for Laryngoscope 125: March 2015
middle ear, mastoid, bony facial nerve canal, petrous apex, and carotid canal involvement. MRI more accurately assesses the parotid region, meninges, and medullary bone.15 With the refinement of imaging technology, CT and MRI offer insight into treatment effect, but clinical improvement is paramount in assessing response.16 Both CT and MRI, however, often remain positive many months to a year after antibiotic treatment is terminated. Gallium 67 citrate scans, especially in combination with single-photon emission computed tomography/ computed tomography, offers the optimal way to follow disease progression/resolution.17
CONCLUSION Although rare, pseudoaneurysms of the ICA require prompt diagnosis and treatment to prevent hemorrhage and death. It should be included in the differential diagnosis of cranial nerve palsies in patients with MOE, as well as patients presenting with a parapharyngeal mass in an infectious setting. In all cases, prompt management, whether endovascular (coiling or stenting) or surgical, is needed to avoid possibly fatal outcomes. Concurrent treatment of the MOE with culture- directed long-term antibiotics is crucial.
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Baker et al.: ICA Pseudoaneurysm in MOE