Pseudoaneurysm of the Internal Carotid Artery After Craniofacial Traumatism: Series of Cases and Integrative Literature Review

Abstract

The present study aims to develop an integrative review about pseudoaneurysms after traumatic facial trauma, through the analysis of its etiology, type of fracture, signs and symptoms, time elapsed between the trauma and the exposure of signs and symptoms, and treatment performed in these cases. Furthermore into the report, there are 3 clinical cases that occurred in the Buccomaxillofacial Surgery and Traumatology service of Hospital da Restauração Recife/PE. The study was carried out in 2 phases, first, a digital research about post-traumatic craniofacial pseudoaneurysm was performed in the following databases “MEDLINE/PubMed,” “Scielo” and “Scopus,” in September 2019, with 5 articles being included. In the second, 3 cases of patients with high-impact facial trauma who developed pseudoaneurysm of the internal carotid artery were reported. The average age of the patients was 35.6 years, all patients were male (100%), the majority (60%) being victims of an automobile accident, 3 patients (60%) suffered craniofacial trauma and 2 (40%) had trauma only to the face, most of them had symptoms later on due to the trauma, in 80% of cases the therapy instituted was embolization. Cases of high-impact craniofacial traumas, which present epistaxis and/or ophthalmological alterations correlated to fractures, should be carefully evaluated, determining a better prognosis for the patient.

Introduction

Craniofacial trauma can result in severe complications such as upper airway involvement, bone fractures and profuse bleeding. Post-traumatic pseudoaneurysms of the intracranial portion of the internal carotid artery (ICA) are uncommon. They represent a rare complication of head trauma, especially in adults. Mortality is high (up to 50%) and these lesions are often located in the cavernous portion of the ICA, however, they can also appear in the petrous, and supraclinoid portions.[1]

In 1971 Maurer described a triad composed of epistaxis, ipsilateral amaurosis and orbital fracture as a pathognomonic sign of internal carotid artery pseudoaneurysm (ICAP), if they occur after craniofacial trauma. However, the presence of any of these signs alone should trigger an investigation for ICAP.[2]

The management of epistaxis in initial care is an important part of the advanced trauma life support (ATLS) since it interferes with the maintenance of the patent’s airway and hemodynamic stability. Overall, severe epistaxis due to facial trauma is not common, however, when it occurs it may compromise the patient’s vital signs.[3]

Some treatment methods have been proposed in the literature for cases of intense nasopharyngeal bleeding that are unresponsive to local measures at the bleeding site such as ligation or clamping of large vessels in the neck, and endovascular procedures for embolization.[4]

Thus, the objective of this work was to perform an integrative review about pseudoaneurysms after craniofacial trauma focusing on the analysis of the etiology, type of fracture, signs and symptoms, time elapsed between the trauma and the appearance of signs and symptoms and the treatment performed. Additionally, we report 3 cases of pseudoaneurysms of the internal carotid artery (ICA) which occurred after craniofacial trauma.

Patients and Methods

Study Design and Integrative Review

This study was performed in 2 steps according to a method previously described.[5,6] In the first step, 2 authors (PSP and RRLS) independently searched in September 2019 for articles about post-traumatic craniofacial pseudoaneurysm in the following databases: “MEDLINE/PubMed,” “Scielo” and “Scopus.” For this search, the following combination using the Boolean operators was used: (aneurysm OR Maurer triad) AND (facial OR maxillofacial OR nasal OR nose OR zygomatic OR malar OR maxilla OR maxillary) AND (trauma OR fracture)/ The inclusion criteria for selecting articles were: full text availability in English, Portuguese or Spanish, reporting post-traumatic pseudoaneurysms in patients with a history of maxillofacial trauma, and being published in the last 10 years.

In the second step, 3 patients with post-traumatic ICAP were reported. Patients were informed about the publication of the cases and signed a consent form.

An integrative review was carried out to guide the answer to the following questions: 1) What is the most frequent type of fracture associated with the occurrence of post-traumatic ICAP? What is the most performed treatment for post-traumatic ICAP?

The articles were initially selected based on their titles and abstracts. After reading the abstracts, the studies that seemed to correspond to the purpose of this review were read in full, and once they met the inclusion criteria, they were included in study. The following data was extracted: location of the research, year of publication, patient’s age and gender, etiology of the trauma, location of the injury, type of fracture, time elapsed between occurrence of trauma and the appearance of signs and symptoms (latency time), and treatment performed.

Data Analysis

The SPSS Software (version 23.0; IBM, Armonk, NY) was used for descriptive statistics.

Results

Integrative Review

The initial screening included 331 articles. The inclusion criteria were applied, and 44 articles were selected based on their title and abstract. After reading the full text, 39 studies were excluded because they did not reported the etiology of trauma neither patients’ signs and symptoms. Therefore, 5 articles were included for analysis and discussion. The search summary can be seen in Figure 1.

The variables evaluated in the selected articles are described in

Table 1. Data of Literature Cases Presenting Pseudoaneurysm of the Internal Carotid Artery After Craniofacial Traumatism.

.

Age

The patients’ age ranged from 22 to 49 years old, with an average of 35.6 (Table 1).

Gender

The sample (Table 1) consisted of 5 male individuals (100%)

Etiology

Most patients were involved in vehicle accidents (n = 3, 60%). One was involved in a fall from a tree (20%), and 1 was related to an aggression by an unknown weapon (20%) (Table 1).

Injury Location

Of the patients, 3 had a skull base fracture (60%), and 2 had skull base fractures and associated facial fractures (40%).

Type of Fracture

One (20%) had a skull base fracture, 1 (20%) had an ethmoid fracture, 1 (20%) had a temporal fracture, 1 (20%) had multiple fractures (frontal, ethmoid, zygoma, orbit, skull base), and 1 (11.1%) the type of fracture was not specified.

Latency Time

Most patients presented symptoms either immediately or late to the trauma (n = 3, 60%), and 40% (n = 2) had late symptoms.

Signs and Symptoms

The most frequent sign in the cases evaluated was epistaxis alone (n = 4, 80%), followed by bleeding related to ophthalmological changes (n = 1, 40%), nasopharyngeal bleeding (n = 1, 10%), and 1 of the studies did not specify signs and symptoms presented (Table 1).

Treatment

Isolated embolization was the most frequently performed treatment in the cases evaluated, corresponding to 80% of all cases (n = 4), and in 1 case (n = 1, 20%) the patient died before treatment.

Case Series

Case 1

A 24-year-old male patient was involved in a vehicle accident and was admitted to Hospital da Restauração Governador Paulo Guerra (Recife, Brazil). On physical examination, he was conscious, oriented and showed active epistaxis and diffuse facial edema, periorbital ecchymosis, and a complaint of blurred vision in the right eye. Computed tomography (CT) showed a fracture of the right zygomatic-orbital complex and a naso-orbital ethmoid fracture (NOE) (Figure 2). In the initial care, anterior nasal packing was performed to control bleeding and was maintained for 48 hours. After this period, it was removed and patient was observed for new episodes of nasal bleeding. After a week of hospitalization, the patient underwent a surgical procedure to treat the facial fractures. No immediate complication was observed. He was discharged from the hospital 48 hours later and his return was scheduled for the next 30 days. On his return, the blurred vision complaint remained and his was referred to an ophthalmological consultation. After 60 days of the accident, the patient returned to the emergency room with active and intense epistaxis. Angiography was requested and a large pseudoaneurysm was detected in the cavernous segment of the internal carotid artery with its largest diameter being 16.5 × 9.7 mm (Figure 3). Therefore, it was decided to occlude the parent vessel. A microcatheter was used to perform embolization with a nitinol coil through the femoral route under general anesthesia. The final angiographic control showed symmetry in arterial and venous times in both brain hemispheres. The patient was discharged from the hospital 48 hours after the endovascular procedure. The patient did not complain of further episodes of bleeding on his 18-month follow-up consult.

Case 2

A 24-year-old male patient was involved in a motorcycle accident and was admitted to the emergency room of Hospital da Restauração Governador Paulo Guerra (Recife, Brazil). Upon initial physical examination, he was conscious, eupneic, and did not show signs or symptoms of traumatic brain injury. He had a right periorbital edema and ecchymosis, normal visual acuity and ocular motricity, and a satisfactory mouth opening. Skull and facial CT scans were requested, in which neurological lesions were ruled out, however, there was a fracture of the right orbital zygomatic complex without significant displacement (Figure 4). A conservative treatment was chosen because there was no clinical or functional limitation. The patient was discharged from the hospital with an outpatient appointment scheduled for a 15, 30 and 60-day follow-up. He returned in the first 2 follow-ups and showed a good projection of the right zygomatic, a satisfactory mouth opening. He had no visual or other complaints. After 2 months, he was admitted to the emergency room with active epistaxis. He reported previous episodes of bleeding and a history of new trauma. An anterior nasal packing was performed to control hemorrhage. Angiography was requested to rule out possible vascular injury. A massive epistaxis while waiting for the procedure occurred and posterior nasal packing was performed. Blood transfusion was initiated for volume replacement. The bleeding was stabilized, and the patient was taken with a lower level of consciousness to undergo the examination under general anesthesia. An internal carotid artery pseudoaneurysm was evident (Figure 5) and subsequent embolization with a nitinol coils was performed. The patient remained under observation in the ward for 72 hours, and no further bleeding episodes occurred. He did not report new bleeding episode on his 18-month follow-up visit.

Case 3

A 34-year-old male patient was admitted to the emergency room of the Hospital da Restauração Governador Paulo Guerra (Recife, Brazil) with a history of motorcycle accident. He showed massive epistaxis, and an anterior nasal packing was performed immediately after his admission to maintain his airway and stabilize bleeding. After 48 hours of admission to the service, the patient was unplugged and once stable, he went to the ward to correct post-traumatic anemia and later elective surgery to fix a fracture of the right orbital zygomatic complex (Figure 6). Clinically, he had amaurosis in his right eye (being monitored jointly by the ophthalmology team) and had a complaint of nasal obstruction. Thirty days after admission and just about to be discharged from hospital, he presented intense nasopharyngeal bleeding and rapidly evolved to a decreased level of consciousness, hypovolemic shock and cardiorespiratory arrest. Cardiopulmonary resuscitation and intubation were performed. A nasal packing was placed and a large pseudoaneurysm of the internal carotid artery was found on an arteriogram (Figure 7). The patient was embolized with nitinol coils, under general anesthesia and through the femoral route. After the procedure, the nasal packing was removed, and no new episodes of bleeding were noticed. The patient was discharged 1 week after embolization. In the 24-month follow-up visit, the patient did not report further episodes of bleeding.

Discussion

This review confirmed that the occurrence of ICA pseudoaneurysm after craniofacial trauma is a rare event reported in the surgical literature. In addition, it also confirmed that endovascular therapy was the most performed option to treat these cases.

According to the present review, most cases of ICA pseudoaneurysms were associated with vehicle accidents and males were most affected. It is believed that the incidence of this condition is associated with the fact that this gender is epidemiologically more involved in this type of trauma.[12]

The development of an aneurysm is determined by the degree of vascular damage, blood flow and the elasticity of neighboring tissues. In regard to the previous 2 factors, there is less chance of a platelet plug controlling bleeding in larger arteries.

The fibrous tissue surrounding the blood vessel is elastic and has a compressive effect that helps to control bleeding and leads to the formation of a hematoma.[13] When the blood flow between the arterial laceration and the hematoma is maintained, a pseudoaneurysm develops. If the leak persists, the center liquefies, which produces a cavity lined with tissues that resemble endothelial cells.[14] The vessel has a pseudo-layer that surrounds it, maintaining its continuity with the lumen and leading to the gradual expansion of the aneurysm, which progressively increases and ruptures.[13]

This study revealed that in the last 10 years no ICA pseudoaneurysms were reported after isolated facial trauma, all cases reported were after isolated head (60%) or craniofacial (40%) trauma. However, the cases reported here showed the occurrence of pseudoaneurysms in patients who had fractures of the middle third of the face, without association with simultaneous cranial fractures.

Trauma victims who have severe and late bleeding should be investigated for the presence of pseudoaneurysms. In some cases, however, the initial angiography may not reveal this alteration, which is likely related to a bone fragment obliterating the carotid artery, retaining the blood and consequently interfering with the flow. Over time, due to the pulsatility of the vessels the pseudoaneurysm ruptures.[15] Ruptures may also occur if the systolic pressure exceeds the resistance of neighboring soft tissues, which leads to life-threatening bleeding. The rarity of the pseudoaneurysm in the head region is due to the complete transection of the vessels usually occurring more often than a partial laceration. In addition, larger and deeper vessels are protected from injuries caused by the underlying soft tissues.[16] For this reason, if bleeding persists, the angiography should be repeated for further investigation. As observed in all the 3 reported cases, the initial images did not suggest the existence of a pseudoaneurysm, and a second investigations was required.

There is a direct communication between the vessel lumen and the pseudoaneurysm. The structure of the pseudoaneurysm is formed by fibrous tissue, while a true aneurysm has an endothelial wall. For this reason, there is a greater chance of rupture of a pseudoaneurysm when compared to an aneurysm of the same size.[8]

Of the studies reviewed, 2 cases had late episodes of severe epistaxis, and the other 3 had episodes of immediate and late epistaxis. In the reports reported in our institution, we observed 2 cases of late epistaxis and 1 case in which there was 1 immediate and 1 late bleeding event.

The ophthalmological alterations presented in some cases is related to the impairment of the blood and nervous structures of the ocular region (i.e. abducens and the optic nerve) which is closely related to the cavernous sinus. Usually, these changes occur unilaterally and ipsilaterally, however, contralateral or bilateral involvement may occur due to the anatomical anastomosis of the 2 cavernous sinuses. In this review, we found only 1 report of ophthalmological alterations related to post-traumatic pseudoaneurysm.[7] In the cases presented by us, only 1 of the 3 cases showed these alterations.

When epistaxis occurs, local measures must be taken to avoid volume loss and consequently hemodynamic instability. These measures include: anterior or posterior nasal packing, compression, cauterization and bandages. If symptoms persist, it is imperative to investigate and rule out the presence of pseudoaneurysms and perform proper treatment, either by ligation of large vessels or by endovascular therapy.[17,18]

Endovascular therapy has shown promise in the treatment of pseudoaneurysms when compared to vessel ligation, considering that embolization is less invasive, and is associated with less morbidity and mortality. The literature indicates a success rate of around 96%, and complications were reported in 1.9% to 21% of cases.[3,4] All cases included in this review were submitted to endovascular therapy, with the exception of 1 report where the patient died before treatment. In addition, all cases reported by us also had endovascular therapy as the treatment of choice.

Given the experience of our service, trauma patients who have persistent epistaxis associated with a mid-face fracture must be followed for a long period. It is recommended that they are submitted to an annual routine exam, which includes an angiotomography to detect pseudoaneurysms. Therefore, early treatment may be performed, and morbidity and mortality are reduced.

In conclusion, cases of high-impact craniofacial trauma that present epistaxis and/or ophthalmological alterations and facial fractures should be evaluated with caution, from initial care to long-term follow-up, since early diagnosis of an ICA pseudoaneurysm will allow better management of the condition.