Recurrent carotid blowout syndrome: diagnostic and therapeutic challenges in a newly recognized subgroup of patients

Abstract

Background and purpose: To our knowledge, recurrent carotid blowout syndrome (rCBS) has not been well described. Our purpose was to review our institution's recent experience with patients who presented with multiple episodes of carotid blowout syndrome (CBS), and who were referred for emergent diagnostic angiography and endovascular therapy.

Methods: We retrospectively reviewed the last 46 consecutive patients who had a clinical diagnosis of CBS. All patients were examined and treated prospectively according to a standardized protocol. Most patients (43 of 46) had undergone extensive primary and salvage radical surgery with intraoperative brachytherapy or external beam radiation or both. The remaining three patients had either traumatic or iatrogenic CBS.

Results: Twelve patients (26%) in our series had more than one episode of CBS in which a total of 32 (20 recurrent) events were observed (average 2.7, range 2-4). Intervals of rCBS ranged from 1 day to 6 years. Thirteen (65%) of 20 recurrent events were attributed to progressive disease (PD), and seven (35%) of 20 to treatment failures (TFs). In the PD group, seven (54%) of 13 had recurrent ipsilateral disease, and six (46%) of 13 had recurrent contralateral disease. Etiologies of rCBS were as follows: seven exposed carotids; seven carotid pseudoaneurysms; eight small-branch pseudoaneurysms; five tumor hemorrhages; three hyperemic/ulcerated wounds; and one aortic arch rupture. Twenty-seven of 32 events were treated with endovascular therapy, which included the following: nine carotid occlusions; 11 small-branch embolizations; three transarterial tumor embolizations; one carotid stent; and two direct-puncture embolizations. Four of six TFs were retreated successfully with endovascular therapy; the remaining two TFs were managed successfully by surgery. In the PD group, hemorrhagic complications of rCBS were managed successfully in all but one patient, who died. No permanent neurologic or ophthalmologic complications occurred.

Conclusion: Recurrent CBS is a frequently encountered problem in which most cases are caused by PD resulting from both multifocal iatrogenic arteriopathy and occasional wound complications that are characteristic of aggressively managed head and neck surgical patients. Initial TFs are encountered often as well. Despite the diagnostic and therapeutic challenges of rCBS, most cases can be retreated effectively.

fig 1.

Illustrative case 1 of the spectrum of rCBS and its management. A and B, Forty-six-year-old woman (patient TL) initially presents with CBS (Group 3). Lateral view from left common carotid injection (A) shows a pseudoaneurysm (arrow), which successfully was treated with therapeutic balloon occlusion (B). C and D, Six years later, the patient develops a second episode of CBS. Lateral view from right external carotid injection (C) shows a hypervascular tumor of the oropharynx and hypopharynx (arrows) that is responsible for bleeding. Lateral view from superselective injection of the ascending palatine artery (D) shows significant supply to the tumor (arrows), which successfully was embolized with PVA. E, Five days later, the patient develops a third episode of CBS, related to recurrent tumor hemorrhage. She was taken to the operating room in which, after induction of general anesthesia and oral retraction, the tumor was punctured under direct visualization and fluoroscopic guidance with a 23-gauge Chiba needle. Lateral fluoroscopic spot film shows the needle coursing through the tumor (long arrow) with extensive filling of neovasculature upon injection of absolute ethanol mixed with metrizamide (small arrows).

fig 2.

Illustrative case 2 of the spectrum of rCBS and its management. A and B, Sixty-six-year-old woman (patient CH) initially presents with CBS (Group 3). Lateral views from superselective injection of a facial arterial branch (short arrow) in early (A) and late arterial phases (B) show a ruptured pseudoaneurysm with extensive extravasation (long arrows). This successfully was treated with coil embolization. C, Twenty-one days later, the patient develops a second episode of CBS (Group 1) due to a flap dehiscence. Oblique view from right CCA injection shows no evidence of pseudoaneurysm and prior ligation of the ECA. The patient failed BTO at this time, prompting a flap revision. D, Seventeen days later, the patient develops a third episode of CBS (Group 3). Oblique view from right CCA injection shows a large pseudoaneurysm of that vessel. Acute hemorrhage initially was arrested by placement of two overlapping 8 × 20-mm Wallstents across the rent of the artery (not shown). E and F, One day later, the patient develops a fourth episode of CBS (Group 3) due to a TF of the previously deployed stents. After inflation of a balloon occlusion catheter across the carotid rent, the pseudoaneurysm was directly punctured and embolized with cyanoacrylate. Fluoroscopic spot film (E) and subtracted-control angiography (F) from right CCA injection (oblique view) shows complete obliteration of the pseudoaneurysm with cyanoacrylate (arrow) and patency of the parent artery.