Endovascular treatment of the subclavian artery pseudoaneurysm: Two case reports

Hao Cai; Yunfeng Pang; Ziqiang Sun; Song Jin

doi:10.4103/2589-9686.377611

CASE REPORT

Year : 2023 | Volume : 6 | Issue : 1 | Page : 20-24

Endovascular treatment of the subclavian artery pseudoaneurysm: Two case reports

Hao Cai, Yunfeng Pang, Ziqiang Sun, Song Jin
Department of Vascular Surgery, Affiliated Hospital of Jining Medical University, Jining, China

Date of Submission	05-Oct-2022
Date of Decision	22-Feb-2023
Date of Acceptance	24-Feb-2023
Date of Web Publication	26-May-2023

Correspondence Address:
Dr. Song Jin
Department of Vascular Surgery, Affiliated Hospital of Jining Medical University, No. 89, Guhuai Road, Jining 272029
China

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/2589-9686.377611

Abstract

Endovascular treatment is widely used for treating the subclavian artery pseudoaneurysm (SAP) because of its small trauma, fast recovery, exact effect, and low incidence of complications. However, when the systemic disease or infection that causes SAP is in the active phase, treatment of the primary disease is more critical. Surgical intervention should be performed after systemic symptoms have been controlled unless the situation is particularly critical. We introduced two cases of SAP caused by brucellosis and Behcet's disease and discussed the course of treatment.

Keywords: Behcet's disease, brucellosis, endovascular treatment, subclavian artery pseudoaneurysm

How to cite this article:
Cai H, Pang Y, Sun Z, Jin S. Endovascular treatment of the subclavian artery pseudoaneurysm: Two case reports. Vasc Invest Ther 2023;6:20-4

How to cite this URL:
Cai H, Pang Y, Sun Z, Jin S. Endovascular treatment of the subclavian artery pseudoaneurysm: Two case reports. Vasc Invest Ther [serial online] 2023 [cited 2023 Jun 8];6:20-4. Available from: https://www.vitonline.org/text.asp?2023/6/1/20/377611

Introduction

Subclavian artery pseudoaneurysm (SAP) is a rare disease with an incidence of approximately 1%–2%. Common causes of SAP are trauma (fractures and iatrogenic injuries), atherosclerosis, thoracic outlet syndrome, systemic diseases, and infections. Iatrogenic injury approximately accounts for 1/3 of trauma cases and occurs during central venous catheterization, dialysis catheter placement, or other medical procedures involving artery puncture of the carotid and subclavian arteries.^[1] SAP due to Brucellosis and Behcet's disease (BD) is extremely uncommon. We reported two cases of SAP and discussed the course of treatment.

Case Reports

Case report 1

A 30-year-old Chinese man was admitted to the hospital with persistent dull pain in the chest and discomfort in the left shoulder for 1 day. He had a history of intermittent oral and perineal ulcers for 5 years. On physical examination, the patient presented with oral ulcers, genital ulcers, and deep tenderness in the left supraclavicular fossa. Computed tomography angiography (CTA) demonstrated a pseudoaneurysm with a maximal diameter of 48 mm in the initial segment of the left subclavian artery and a pseudoaneurysm with thrombosis in the distal abdominal aortic [Figure 1]. On additional laboratory tests, the erythrocyte sedimentation rate (ESR) was 55 mm/H, the C-reactive protein (CRP) level was 90.8 mg/L, the interleukin-6 (IL-6) was 64.9 pg/ml, and the immunoglobulin A (IgA) was 4.3 g/l. The patient was diagnosed with BD by a rheumatology consultation.

Figure 1: (a) CTA demonstrating a pseudoaneurysm of the initial segment of the left subclavian artery (black arrow), (b) CTA demonstrating an aneurysm of the distal abdominal aorta with thrombosis (black arrow). CTA: Computed tomography angiography

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Surgery or endovascular treatment can cause complications because of the active phase of BD and the strong vascular inflammatory response. Hence, a combination of medical therapy with 400 mg methylprednisolone once a day injected and 200 mg compound cyclophosphamide tablets once every other day orally was started. After his condition improved, the patient was discharged and continued to receive hormones and immunosuppressive drugs.

Two weeks later, the patient was readmitted to the hospital due to sudden pain in the chest and left shoulder for 1 day. On physical examination, the patient presented with a deep tenderness in the left supraclavicular fossa and ptosis of the left eyelid. CTA demonstrated a more advanced pseudoaneurysm of the left subclavian artery affecting the left vertebral artery with a maximal diameter of 69 mm. On additional laboratory tests, the ESR was 16 mm/H, and the CRP level was 54.3 mg/L.

Given the high risk of SAP rupture and the improvement of BD symptoms, the patient received endovascular treatment. Digital subtraction angiography (DSA) showed a pseudoaneurysm in the distal abdominal aortic [Figure 2]a. A 24 mm × 80 mm covered stent (Lifetech, Ankura, CHINA) was implanted through the right common femoral artery to isolate the abdominal aorta pseudoaneurysm [Figure 2]b. The 8 mm × 50 mm, 9 mm × 50 mm, and 10 mm × 50 mm covered stents (Gore Viabahn, USA) were implanted from distal to proximal in the left subclavian artery through the right common femoral artery approach [Figure 2c]. Angiography showed no internal leakage and the left subclavian artery was unobstructed [Figure 2d]. The patient was discharged and continued receiving prednisone acetate tablets, aspirin, and clopidogrel. CTA demonstrated the subclavian artery and abdominal aorta were unobstructed at 3 months of operation. The patient did not return for follow-up due to the COVID-19 pandemic.

Figure 2: (a) DSA demonstrating a pseudoaneurysm in the distal abdominal aortic (white arrow), (b) DSA demonstrating abdominal aortic stent was unobstructed without internal leakage, (c) DSA demonstrating pseudoaneurysm of the left subclavian artery (white arrow), (d) DSA demonstrating subclavian artery stent was unobstructed without internal leakage. DSA: Digital subtraction angiography

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Two years later, the patient was readmitted for pain in the right lower abdomen for 8 h. On physical examination, the patient presented with a scrotal ulcer. CTA demonstrated an aneurysm between the lower end of the abdominal aortic stent and aortic bifurcation [Figure 3]a. On additional laboratory tests, the ESR was 36 mm/H, the CRP level was 65.0 mg/L, the IL-6 was 49.1 pg/ml, and the IgA was 4.1 g/L. Patient's condition gradually stabilized after hormone and immunosuppressive treatment. Considering the risk of rupture due to the irregular shape of the aneurysm, the patient received endovascular treatment again. DSA showed an aneurysm between the lower end of the abdominal aortic stent and aortic bifurcation [Figure 3]b. A unibody stent graft (Microport, Aegis-B, CHINA) was implanted to isolate the abdominal aneurysm through the bilateral common femoral artery approach. Angiography showed the aneurysm disappeared, and the stent was unobstructed. The patient was discharged and continued receiving hormones and immunosuppressive drugs. At the 1^st month follow-up, CTA demonstrated abdominal aortic stent was unobstructed [Figure 3]c. At the 3^rd month follow-up, the patient's laboratory test results were normal except for high white blood cells. The patient was asymptomatic and continued receiving hormones and immunosuppressive drugs.

Figure 3: (a) CTA demonstrating a pseudoaneurysm in the initial segment of the right subclavian artery (white arrow), (b) DSA demonstrating an aneurysm between the lower end of the abdominal aortic stent and aortic bifurcation (white arrow), (c) CTA demonstrating abdominal aortic stent was unobstructed at the 1^st month follow-up. CTA: Computed tomography angiography, DSA: Digital subtraction angiography

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Case report 2

A 70-year-old Chinese man was admitted to the hospital with hemoptysis for 8 days and back pain for 3 days. On physical examination, the patient has no apparent abnormalities. CTA demonstrated a pseudoaneurysm in the initial segment of the right subclavian artery [Figure 4]. On additional laboratory tests, the CRP level was 82.2 mg/L. Complement, Ig, rheumatoid factor, and tumor markers were normal. After admission, the patient had recurrent fever with a maximum temperature of 39°C, as well as cough, blood in the sputum, and night sweats. A positive Brucella agglutination test was performed, and brucellosis was diagnosed. Minocycline and rifampin were given to treat the infection of brucellosis.

Figure 4: (a) Coronal plane imaging of CTA demonstrating a pseudoaneurysm in the right subclavian artery (white arrow), (b) Cross-sectional imaging of CTA demonstrating a pseudoaneurysm in the right subclavian artery (white arrow). CTA: Computed tomography angiography

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Given the high risk of SAP rupture, the patient received endovascular treatment after his temperature was normal. DSA showed a pseudoaneurysm of the right subclavian artery [Figure 5]a. The 9 mm × 50 mm and 13 mm × 50 mm stents (Gore Viabahn, USA) were implanted from the brachiocephalic trunk to the right common carotid artery. The proximal part of the right subclavian artery was embolized with two 20 mm × 40 mm Interlocks (Boston Scientific, USA). DSA showed that the right common carotid artery was unobstructed, and the SAP disappeared [Figure 5]b. The patient was discharged and continued receiving minocycline and rifampin.

Figure 5: (a) DSA demonstrating pseudoaneurysm of the right subclavian artery (white arrow), (b) DSA demonstrating that the proximal pseudoaneurysm had disappeared. DSA: Digital subtraction angiography

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At the 3^rd month follow-up, the patient had no other fever but was still expectorated with bloody sputum. On additional laboratory tests, the Brucella agglutination test was still positive. The patient continued receiving minocycline and rifampin. At the 12^th month telephone follow-up, the patient indicated no further expectorating with bloody sputum.

Discussion

The incidence of SAP is rare. The most common cause of SAP is trauma, and iatrogenic injuries account for about 1/3 of trauma cases. There are few reports on SAP caused by BD or brucellosis. Most pseudoaneurysms occur in the initial segment of the subclavian artery. Common symptoms of SAP include cephalgia, dorsalgia and arm pain, Horner syndrome, hoarseness, difficulty swallowing, arm ischemia, rupture or distal embolism of SAP, and congestion of arm veins. The most common clinical manifestations of pseudoaneurysm rupture are pulsing masses and bleeding, whereas symptoms due to neurological impairment of the arm are the second most common primary complaints. Other symptoms include respiratory symptoms (cough, dyspnea, and hemoptysis), pain, and hypovolemic shock.^[2]

Imaging tests for SAP include ultrasound (US), CTA, and DSA. US can only provide a preliminary clinical impression because it can not show pleural cavity, mediastinum, lung, and bone structures.^[3] CTA can rapidly make an accurate diagnosis and rule out other problems. DSA is generally used for preoperative angiography in patients with opportunities to undergo endovascular treatment.

BD is a vasculitis of variable vessels that commonly appears in young adults aged 20–40. Common symptoms of BD include recurrent oral sores, vaginal ulcers, uveitis, and skin lesions.^[4] Vasculo-BD appears in only 2%–7% of patients, including arteriovenous thrombosis and aneurysms.^[5] Aneurysms caused by BD have been found to affect the pulmonary artery, abdominal aorta, and subclavian artery.^[4],[5],[6] For small asymptomatic aneurysms with a low risk of rupture, high-dose hormonal and immunosuppressive therapy may be sufficient. Cyclophosphamide may be given as monthly intravenous pulses, and glucocorticoids are usually given as three consecutive intravenous methylprednisolone pulses followed by oral prednisolone (or prednisone) at a dose of 1 mg/kg/day.^[7] For patients at high risk of ruptured aneurysms, surgical treatment should be given priority. However, patients frequently experience postoperative problems such as graft blockage or the creation of anatomical pseudoaneurysms if they are in the active phase of BD.^[8] Therefore, medical treatment needs to be administered throughout treatment.

Brucellosis is a commonly encountered zoonotic disease with over half a million new cases annually and prevalence rates in some countries exceeding ten cases per 100 000 population. Humans become infected with the disease through ingesting contaminated milk products or meat or direct contact with the mucous membranes.^[9] The main clinical manifestations include undulant fever, arthralgia, and fatigue. Brucella can infect endothelial cells and induce a severe inflammatory response that may lead to cardiovascular complications. Cardiovascular complications due to brucellosis are rare but can cause serious complications, including infective endocarditis and aneurysms.^[10] Infectious aneurysms caused by brucellosis have been found to affect the coronary artery, abdominal aorta, cerebral artery, and subclavian artery.^{[10],[11],[12],[13]} Treatments of Brucella-induced pseudoaneurysm involve open surgery or endovascular treatment together with long-term anti-brucellosis treatment. Rifampin, doxycycline, and streptomycin are the basic clinical drugs used for anti-brucellosis treatment.^[14] The traditional surgical modality is resection of the infected tissue and revascularization.^[7] Endovascular treatment is less invasive and can reduce mortality compared to traditional open surgery.^[8] Given these factors, we used medication and endovascular surgery in our case.

Surgical treatments for SAP include open surgery, endovascular treatment, and US-guided thrombin injection. Open surgery includes aneurysmorrhaphy, ligation, and bypass. Due to the subclavian artery's anatomical characteristics, open surgical techniques can cause injury to the brachial plexus or adjacent blood vessels, increasing morbidity and mortality. A subclavian or supraclavicular incision can be used when the intrathoracic segment of SAP is not impacted; otherwise, a midsternal or left extrathoracic incision should be required.^[2] Endovascular treatment approaches include covered stent placement and coil embolization. The mean technical success rate of endovascular repair was 96.9%.^[15] To obtain an adequate anchoring area, the covered stent may cover the vertebral artery and lead to stroke or other complications. Therefore, intraoperative preparation for vertebral artery revascularization should be performed. Vertebral artery revascularization should be considered if these symptoms occur.^[16] SAP can be effectively treated with thrombin injections guided by ultrasonography.^[17] The most common complication is thromboembolism. Thrombin injection therapy must be done with caution. The needle tip should be placed in the pseudoaneurysm, far from the neck of pseudoaneurysm, and the amount of thrombin should not be excessive. Endovascular treatment of SAP due to BD or brucellosis is safe and effective with good short-term results. However, anti-infective therapy or immunotherapy should be administered throughout treatment. Further studies are needed to determine its long-term efficacy and patency.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Talent Development Program of Affiliated Hospital of Jining Medical University (Project ID: jyfy-yc-001).

Conflicts of interest

There are no conflicts of interest.

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