
==== Front
Int J Surg Case Rep
Int J Surg Case Rep
International Journal of Surgery Case Reports
2210-2612
Elsevier

S2210-2612(24)01070-8
10.1016/j.ijscr.2024.110289
110289
Case Series
Is there an association between arteriovenous fistulas and axillary artery aneurysms? A report of two cases
Abdulrahim Omer a
Khalil Bassam Abdulhailm a
Alenazi Shahad Farhan a
Alanezi Tariq alanezitariq@gmail.com
b⁎
Al-Omran Mohammed acde
a Department of Surgery, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia
b College of Medicine, King Saud University, 11322 Riyadh, Saudi Arabia
c Division of Vascular Surgery, St. Michael's Hospital, Unity Health Toronto, Toronto, Ontario, Canada
d Department of Surgery, University of Toronto, Canada
e Li Ka Shing Knowledge Institute, St. Michael's Hospital, Unity Health Toronto, Toronto, Ontario, Canada
⁎ Corresponding author. alanezitariq@gmail.com
14 9 2024
10 2024
14 9 2024
123 11028929 7 2024
5 9 2024
9 9 2024
© 2024 The Authors
2024
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction

Axillary artery aneurysms are rare vascular conditions that can present with various clinical manifestations, including neurological deficits and vascular compromise. While the underlying pathophysiology remains complex and multifactorial, potential associations with trauma, arteriovenous fistula formation, and atherosclerosis have been reported.

Presentation Of Case.

Two male patients, aged 33 and 38, with a history of kidney transplantation and previous arteriovenous fistula (AVF) presented with symptoms of upper limb ischemia and neurological compromise. Imaging revealed large axillary artery aneurysms. Open surgical repair was performed for both cases. Two weeks after discharge, one patient showed good pronation and supination with mildly limited extension. The other patient's wrist drop gradually improved with physiotherapy.

Discussion

Multifactorial pathophysiology encompassed altered blood flow dynamics, inflammation, and the underlying vascular pathology. Chief complaints and prior vascular interventions contributed. Open surgical repair was preferred to endovascular approaches, achieving favorable outcomes.

Conclusion

Axillary artery aneurysms in patients with a history of AVF are rare but potentially serious complications. The cases highlight the complexity of axillary artery aneurysms and the need for careful evaluation and surgical intervention This strategy is crucial to prevent potential complications and optimize patient outcomes. Further research is needed to elucidate the precise pathophysiology and the potential association between AVF and the subsequent development of axillary artery aneurysms. Increasing awareness among surgeons could enable earlier detection of aneurysms, thereby reducing the risk of complications.

Highlights

• A report of axillary artery aneurysms in patients with brachiocephalic arteriovenous fistulas (AVFs).

• The study identifies a potential link between AVF and the subsequent development of proximal aneurysms.

• The pathophysiology is complex and involves alterations in blood flow dynamics and other contributing factors.

• Successful surgical treatment of axillary artery aneurysms through open methods is reported.

Keywords

Axillary artery aneurysms
Brachiocephalic arteriovenous fistula
Case series
Kidney transplantation
Open vascular surgery
Arteriovenous fistula
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pmc1 Introduction

Upper limb aneurysms can arise from various factors, including trauma, mycotic lesions, atherosclerotic changes, fistula formation, or post-obstructive lesions in thoracic outlet syndrome [1]. Congenital conditions, viral diseases, or Marfan syndrome are also described [2]. Among upper limb aneurysms, those affecting axillary arteries are often associated with recurrent damage. These aneurysms are frequently associated with recurrent vascular damage, especially in individuals who use crutches or engage in repetitive overhead activities such as baseball pitching [3]. However, axillary aneurysms resulting from arteriovenous malformations have also been reported [4]. Additionally, cases of aneurysm have been described in patients who previously underwent brachial artery to cephalic vein arteriovenous fistula (AVF)1 for hemodialysis [[5], [6], [7]].

Clinically, axillary aneurysms may present as a pulsatile mass, vascular insufficiency, or neurological complications, owing to their proximity to the brachial plexus. Nevertheless, most cases remain asymptomatic until complications such as distal embolization or rupture manifest [8].

Typical management strategies require the evaluation of the underlying pathophysiology, complete clinical and instrumental diagnosis, tailored surgical approaches, and scheduled postoperative assessments including rehabilitation therapy when needed. Within the care pathway, treatment options include open or endovascular methods. The former may be preferred as it can alleviate nerve entrapment caused by physical compression and the inflammatory changes associated with aneurysm development. Therefore, this surgical approach can preserve the patient's functionality and limit the development of neurological sequelae [9].

In this report, we describe two cases of axillary artery aneurysms in kidney-transplanted patients with a previous ipsilateral AVF. The aim is to underline that a careful perioperative care process can allow optimal management and improve outcomes. The patients provided written informed consent to report their case details and images. The work has been reported following the Preferred Reporting of Case Series in Surgery (PROCESS) criteria [10].

2 Case presentation

2.1 Case 1

A 33-year-old man, who had undergone liver and kidney transplants more than 15 years prior, was receiving hemodialysis via a left radio-cephalic AVF. The fistula was ligated due to thrombosis, followed by the creation of a brachiocephalic fistula. Unfortunately, approximately four years later, aneurysmal development necessitated the ligation of this AVF.

The patient presented to our emergency department (ED) with a one-week history of left upper limb numbness, accompanied by a skin rash and discoloration. Moreover, there was delayed capillary refill, warmth in the left hand, and pain in the distal fingers. Upon examination, he was stable with palpable left axillary and brachial arteries, a monophasic signal on the radial artery, and a biphasic signal on the ulnar artery were appreciated. Ultrasound (US) Doppler additionally revealed absent flow in the mid-portion of the ulnar artery, with patent flow distally. The Computed Tomography Angiography (CTA) indicated a partially thrombosed aneurysmal dilatation of the left axillary artery with peripheral calcification, measuring approximately 3.7 cm (Fig. 1). The imaging also demonstrated a completely thrombosed aneurysm in the left distal brachial/proximal radial artery, measuring 2.1 cm. Concerning antiplatelet and anticoagulant therapy, we prescribed apixaban and aspirin due to the distal embolization/occlusion from the aneurysm.Fig. 1 The Computed Tomography Angiography revealed a partially thrombosed aneurysmal enlargement of the left axillary artery with peripheral calcification, measuring approximately 3.7 cm in size (yellow arrow). (For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.)

Fig. 1

The patient was admitted for surgical intervention secondary to left upper limb ischemia, microthrombi, and thrombosed aneurysm of the left brachial artery. A lower extremity saphenous vein mapping revealed the small caliber of the bilateral saphenous veins, indicating a potential for size mismatch. Consequently, the basilic vein was scanned intraoperatively with duplex ultrasound and marked for harvesting. An infraclavicular inverted hockey stick incision was performed (Fig. 2).Fig. 2 Infraclavicular inverted hockey stick incision.

Fig. 2

The left axillary artery was controlled proximally, and dissection was executed. Both the axillary and brachial plexuses were identified and preserved. The dissection was continued in the caudal position, gaining distal control over the left axillary artery distal to the aneurysm. A lazy S-shaped incision was made in the antecubital fossa, exposing the brachial artery (Fig. 3a). Proximal control over the brachial artery and distal control over the radial and ulnar arteries were achieved (Fig. 3b). The basilic vein dissection and mobilization were performed. It was of good size and was harvested and inflated using heparin-saline. The axillary aneurysm was then excised, and an interposition graft was placed using a reversed basilic vein (Fig. 3c).Fig. 3 Surgical approach. The surgical procedure involved control and dissection of the left axillary artery, identification, and preservation of both the axillary and brachial plexuses, followed by continued dissection to gain distal control over the artery. An incision was made in the antecubital fossa to expose the brachial artery (a), with proximal control over it and distal control over the radial and ulnar arteries (b). The basilic vein was dissected, mobilized, and harvested for use, and the axillary aneurysm was removed (c).

Fig. 3

The distal brachial artery was then identified. Furthermore, thrombectomy for the brachial, radial, and ulnar arteries using Fogarty's catheter sizes 3 and 2 was performed due to a thrombosed aneurysm.

Subsequently, a patch angioplasty using the remaining basilic vein was executed for the affected segment of the brachial artery. Finally, the vein graft and the distal radial and ulnar arteries were confirmed to have strong pulsation.

On postoperative day 10, the patient developed swelling and pain at the site of the surgery. Since the US showed a subcutaneous hematoma measuring 9.4 × 3.3 × 2.3 cm, evacuation was performed. The patient's condition improved, the wound healed, and he was discharged with pain medication.

The patient was seen in the clinic two weeks after discharge. He had no complaints and demonstrated good pronation and supination but still had limited extension, and the wound had healed completely. Pathology results showed dilated arterial wall calcification, myxoid degeneration, and secondary thrombus.

2.2 Case 2

A 38-year-old man with a known history of focal segmental glomerulosclerosis and a renal transplant presented to the ED with a one-week history of left arm pain, swelling, paresthesia, and weakness. His renal transplant was performed 15 years prior. The patient was maintained on tacrolimus and mycophenolate mofetil, with baseline graft function marked by creatinine levels of 170–200 μmol/L due to chronic allograft rejection, further exacerbated by acute rejection episodes.

The patient had a history of prior vascular events, including an unprovoked lower limb deep vein thrombosis 8 years earlier, treated with apixaban for three months. More recently, he developed a chronic non-occlusive thrombus in the right common femoral vein and was on apixaban 2.5 mg BID at presentation. Moreover, he had undergone brachiocephalic AVF ligation after transplant and had a history of cellulitis at the ligation site with a mega fistula.

Physical examination showed a palpable left axillary swelling. The axillary and brachial pulses were palpable, and there was a noticeable wrist drop with weakness in the left upper limb. Doppler examination revealed biphasic signals over the radial and ulnar arteries. Notably, CTA of the upper extremity showed a large aneurysm of the left axillary artery measuring 9.6 × 11.8 × 9 cm, located inferior to the shoulder joint just below the subscapularis muscle (Fig. 4A-B).Fig. 4 CT angiography in coronal (a) and transverse (b) sections, showing the aneurysm (highlighted by yellow arrows). (For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.)

Fig. 4

Given the risk of impending rupture and the mass effect of the aneurysm on the radial nerve resulting in the wrist drop, the patient was taken for urgent surgical intervention. The procedure involved repairing the left axillary artery aneurysm with graft anastomosis. The median and radial nerves closely adhered to the large aneurysm. Given the large size of the aneurysm and the presence of a contained rupture, the decision was made to open the aneurysm sac and control the bleeding from within. Upon opening the sac, there was a massive hemorrhage, causing hemodynamic instability and hypotension. Although the bleeding was promptly controlled, the patient reached a pre-arrest status. The protocol for patient-blood management was successfully implemented. Due to the hemodynamic instability and large caliber of the left axillary artery, we opted for a polytetrafluoroethylene (PTFE) graft instead of a great saphenous vein graft to avoid any potential mistmatching. Anastomoses were checked proximally and distally for good inflow. In the immediate postoperative period, the patient was monitored in the intensive care unit for 24 h before being transferred to a regular ward. Daily examinations showed a steady improvement in his left upper limb function. The swelling gradually subsided, and there was a noticeable improvement in the paresthesia and weakness. Although the wrist drop persisted, there was a gradual improvement with physiotherapy. Repeat US examinations confirmed good graft patency with normal biphasic signals over the radial and ulnar arteries. There were no signs of graft complications such as thrombosis or infection and the surgical wound healed without any signs of infection or dehiscence. The patient was discharged on postoperative day five with anticoagulation therapy — also considering prior vascular events — and scheduled follow-ups at two weeks and three months. At these visits, significant improvement in symptoms was reported; moreover, physical examination showed further improvement in muscle strength and nerve function of the left upper limb. The wrist drop resolved substantially, and there was no recurrence of swelling or pain.

3 Discussion

Given the rarity of axillary artery aneurysms in the upper limb [[1], [2], [3], [4], [5], [6], [7]], a nuanced understanding of their clinical presentation and optimal treatment modalities is essential. Additionally, the underlying pathophysiology warrants further in-depth investigation. Therefore, although with different features (Table 1), the presented cases underscore the complexity and diversity of these vascular conditions, emphasizing the potential association between AVF and the development of a proximal aneurysm.Table 1 Key features of the two cases.

Table 1Feature	Case 1	Case 2	
Patient Age	33 years	38 years	
Gender	Male	Male	
Medical background	Liver and kidney transplantation, brachiocephalic AVF	Focal segmental glomerulosclerosis, renal transplant, brachiocephalic AVF, previous vascular thrombosis	
Symptoms	Numbness in left upper limb, skin rash, discoloration	Left arm pain, swelling, paresthesia, weakness, wrist drop	
Imaging Findings	Partially thrombosed left axillary artery aneurysm (3.7 cm), thrombosed brachial aneurysm (2.1 cm)	Large left axillary artery aneurysm (9.6 × 11.8 × 9 cm)	
Surgical Intervention	Open surgery: interposition graft using reversed basilic vein	Open surgery: use of a polytetrafluoroethylene graft	
Perioperative Complications	Postoperative subcutaneous hematoma	Pre-arrest hypotension during surgery	
Outcome at 2 Weeks	Good pronation and supination, mildly limited extension	Gradual improvement in wrist drop with physiotherapy	
Follow-up	No complaints, complete recovery	Significant improvement in symptoms, wrist drop substantially resolved	

The proximal aneurysmal dilation adjacent to an AVF has been described in several case reports. Although many of these cases involve the brachial artery [11,12], axillary artery aneurysms in patients who underwent brachiocephalic AVFs are a rare occurrence [[5], [6], [7]]. In the first case, in addition to the dilation of the ipsilateral brachial artery, the patient developed an axillary aneurysm after the ligation of the AVF. Although a direct and widely recognized association between AVF and axillary artery aneurysms has not been established, a potential relationship in certain scenarios is conceivable. For instance, fistulas may alter blood flow dynamics or increase arterial pressure, theoretically contributing to the development or exacerbation of an axillary artery aneurysm in predisposed individuals. Nevertheless, in addition to the mechanical factor, other elements could play a role in the development of the lesion. For example, Sapienza et al. [12] have proposed an inflammatory process that ultimately destroys the elastic lamina. In this perspective, arterial remodeling could extend proximally to the axillary artery due to the action of vasodilatory factors such as nitric oxide [13]. Furthermore, the immunosuppressive and corticosteroid treatment could be an important cofactor, although the pathogenetic correlations are not fully understood [14].

While repeated trauma has been typically described [15], in both our cases, the clinical history did not indicate important traumatic processes. Atherosclerosis could better explain the underlying pathophysiology. Atherosclerotic plaques on the arterial inner wall can potentially lead to dilatation and vessel weakness, ultimately leading to aneurysm development [4,7]. Soylu et al. [16], for instance, reported a case of a bilateral axillary aneurysm in the context of atherosclerotic processes. Consequently, the presence of atherosclerotic pathology should prompt suspicion.

In the second patient, the complex clinical history, including vascular events such as unprovoked deep vein thrombosis, and chronic non-occlusive thrombosis, are suggestive of multiple concurrent factors. The combination of vascular damage, inflammation, altered blood flow due to the ipsilateral AVF, and predisposing factors, such as underlying conditions and pharmacological treatments, has contributed to the development of both atherosclerotic and aneurysmal pathology.

Diagnosing and managing these cases is challenging. In the first case described, the symptoms included numbness in the left upper limb accompanied by a skin rash and discoloration in the same area, indicative of vascular compromise. The other patient presented with pain and paresthesia. The occurrence of neurological symptoms has been previously documented in the literature, as reported in other case studies [17].

Given their potential for severe complications, such as neurovascular compressive symptoms, thromboembolic phenomena, and even deleterious ruptures [18], swift interventions are crucial. Regarding the treatment options for axillary artery aneurysms, both endovascular and open approaches have been discussed in the literature, with the open approach being considered as the standard of care [19]. The choice between these options depends on several factors, including the size and location of the aneurysm, patient comorbidities, and the potential risks and benefits of each approach. Endovascular methods are gaining popularity since they offer a minimally invasive approach with a safer profile. Their risk of mortality and morbidity has been reported to be lower when compared to the open strategy [20]. However, the endovascular approach for larger aneurysms can be challenging. In contrast, the open approach offers better visualization of the aneurysm and surrounding tissues. This allows for complete resection and safe detaching of any adherent tissue to the aneurysm, including nerves. Consequently, while the endovascular approach offers many benefits, including being less invasive and having a quicker recovery time, it is not suitable for all aneurysms, particularly those with complex anatomy or significant nerve involvement. The open surgical approach remains the preferred method in such cases, allowing for complete resection and the preservation of critical structures, as demonstrated in the cases discussed. Specifically, in the present cases, the aneurysms were freed completely from any adherent nerves and tissue, which allowed the patients to gain full functionality. Moreover, when there is significant nerve involvement, as with the radial nerve in Case 2, the open approach enables careful dissection and protection of these structures, minimizing the risk of long-term neurological deficits. This suggests that the open surgical approach may be superior to the endovascular method, particularly in similar cases. This data supports what is reported in the literature, even if large case studies are unavailable, the open approach is generally preferred [17,19]. On the other hand, the endovascular technique has been mainly applied in high-risk patients and for palliative purposes since the less invasive nature of endovascular surgery may reduce the risks associated with anesthesia and large incisions [21]. This technique is also preferred in cases where the aneurysm is smaller and does not involve complex anatomy [20], or for emergent treatment [22]. When the open surgery is implemented, autologous veins or synthetic material can be used. For autologous veins, opting for a thin-walled basilic vein conduit over a saphenous vein can be justified by its ease of access, and more importantly, when the saphenous vein is compromised.

Furthermore, concerning prosthetics, PTFE and other materials are often employed when autologous veins are unavailable or as in our second case, when there are technical challenges related to vessel size or the need to quickly perform the procedure. The choice between a ring-supported or not depends on the anatomical site and the need for structural integrity in the graft. Ring-supported PTFE grafts are typically used in areas where kinking or compression of the graft is a concern, providing added durability and reducing the risk of graft collapse.

Finally, regarding outcomes, the surgeon's ability to maintain complete control over the surgical field translates into a significantly decreased occurrence of complications [20]. For this aim, the surgical approach could be further improved. For example, integrating endovascular control before open surgery can offer a bridge to definitive repair and potentially mitigate extensive hemorrhage, thereby improving patient outcomes.

4 Conclusion

Axillary artery aneurysms in patients with a history of brachiocephalic AVF are rare occurrences, highlighting the need for further research to clarify their precise pathophysiology and to develop preventive strategies. These cases also emphasize the importance of meticulous preoperative evaluation and diagnosis, along with precise and appropriate surgical intervention. As described in our cases, by adhering to these careful measures, patients with axillary artery aneurysms can achieve favorable outcomes, thereby avoiding the potentially serious complications associated with this condition. Further research is needed to dissect the precise pathophysiology and the potential association between AVF and the subsequent development of proximal axillary artery aneurysms. Although a direct and widely recognized association between AVF and the development of axillary artery aneurysms has not been established, they may be related in certain scenarios. Therefore, increasing awareness among surgeons to be vigilant in patients with AVFs, could enable earlier detection of aneurysms, reducing in turn the risk of complications.

CRediT authorship contribution statement

Study concept or design – OA, BAK, SFA, TA, MA.

Data collection – OA, BAK, SFA, TA, MA.

Data interpretation – OA, BAK, SFA, TA, MA.

Literature review – OA, BAK, SFA, TA, MA.

Drafting of the paper – OA, BAK, SFA, TA, MA.

Editing of the paper – OA, BAK, SFA, TA, MA.

Patient perspective and informed consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Guarantor

Dr. Tariq Alanezi accepts all responsibility of this article.

Declaration of competing interest

The authors declare no conflict of interest.

Data availability

The data for this study will be available from the corresponding author upon reasonable request.

Acknowledgments

We would like to thank Faridul Ahsan and Reem Saleh Alrajhi for their contributions in documenting the patients' information.

1 Abbreviations: Arteriovenous fistulas (AVFs) are anomalous connections between arteries and veins. AVFs can be surgically established, arise from congenital or genetic anomalies, or result from iatrogenic injury or trauma. With the exception of surgically created fistulas, these occurrences are exceedingly uncommon; Emergency room (ER); Ultrasound (US); Computed tomography angiography (CTA)
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