True brachial artery aneurysm in a 3-year-old: A case report

Abstract

Introduction

True brachial artery aneurysms (TBAA) in children are exceptionally rare vascular anomalies, with fewer than 20 cases of idiopathic isolated aneurysms reported in children aged between 2 month to 18 in the medical literature. We present a rare case of a true brachial artery aneurysm in a 3-year-old child without any associated risk or systemic disease. This case adds to limited literature on TBAA and provides valuable insight for clinician managing similar case in resource limited setting.

Case presentation

A previously healthy 3-year-old female presented with a two-week history of progressive left upper extremity swelling. Physical examination revealed a 2 × 2 cm pulsatile mass on the medial aspect of the left proximal arm, 8 cm proximal to the medial epicondyle. CT angiography (CTA) confirmed a 1.7 × 1.6 cm saccular aneurysm of the proximal brachial artery. The patient underwent successful aneurysm repair with reconstruction using reversed great saphenous vein interposition (GSV) graft. Post-operative recovery was uneventful, with normal vascular function maintained at 3-month follow-up.

Discussion

This case demonstrates the importance of early recognition and timely surgical management in paediatric arterial aneurysms. A systematic diagnostic approach with ultrasound and CTA enabled precise surgical planning. Histopathology finding support a congenital-idiopathic aneurysm, enriching our understanding of rare paediatric vascular anomalies. Autologous saphenous vein reconstruction was effective, confirming its role as the preferred conduit for paediatric vascular repairs.

Conclusion

Surgical management of paediatric brachial artery aneurysms using autologous vein reconstruction appears safe and effective. Early intervention may prevent potential complications while providing excellent short-term outcomes.

Highlights

• •True idiopathic brachial artery aneurysm in children is extremely rare. Ultrasound is key initial evaluation and screening.
• •Early referral to a vascular centre and timely intervention prevent complications in peripheral artery aneurysms.
• •Autologous vein grafts are the preferred choice for reconstructing paediatric arterial anomalies.

1. Introduction

An aneurysm is a localized arterial dilation caused by vessel wall weakness. True aneurysms involve all three layers, while false ones are walled off by fibrous tissue. They can be congenital or acquired (e.g., mycotic, syphilitic, traumatic) and are classified as central or peripheral based on location [1]. Peripheral arterial aneurysms (PAA) are rare, with upper limb involvement accounting for only 5 % of all PAA. Most are linked to systemic diseases and may indicate diffuse aneurysmal disease [[2], [3], [4]].

True brachial artery aneurysms (TBAA) are extremely rare in children, with reported incidence of less than 0.5 % [5,6]. While most cases are often associated with underlying conditions such as Kawasaki disease, infections, trauma, vasculitis, fibromuscular dysplasia, or connective tissue disorders [1,2,4], isolated TBAA, in otherwise healthy children with no underlying cause, are extremely rare, with fewer than 20 cases reported in children aged 2 months to 18 years [[1], [2], [3], [4],7,8]. TBAAs present as painless masses or with symptoms such as pain, ischemia, or nerve compression and rarely symptomatic embolization. They should be differentiated from pseudo aneurysms, hematomas, vascular malformations and other upper limb lesions [3]. The natural history of untreated brachial artery aneurysms includes risks of thrombosis, distal embolization, rupture and potential limb-threatening neurovascular compromise [1,3,7]. Limited access to advanced imaging and genetic testing poses challenges in diagnosing rare vascular anomalies in resource limited setting. In Ethiopia, limited access to vascular services results in delay to get appropriate management.

We present a rare case of a healthy 3-year-old female child who presented with an idiopathic isolated TBAA, timely diagnosed by a variety of multimodality imaging techniques, and successfully treated at Hawassa University Hospital a tertiary centre with expertise in vascular surgery with autogenous vein grafting. Postoperative outcomes showed no ischemia or neurologic deficits in the upper limb. This case adds to the limited literature on TBAA and provides valuable insights for clinicians in the diagnostic approach, surgical decision-making and postoperative management in resource limited settings.

This case report is reported in line with the SCARE criteria [9].

2. Case presentation

A previously healthy 3-year-old female presented with a two-week history of left upper extremity swelling, initially noticed by her mother during bathing. The swelling had progressively increased in size but was not associated with pain. Initial evaluation at a local hospital included an ultrasound that suggested a proximal brachial artery aneurysm, prompting referral to our tertiary centre for specialized vascular management at Hawassa University Hospital, Ethiopia. She had no history of fever or systemic infection, numbness, trauma, connective tissue disorder, or family history of vascular anomalies. At presentation, the patient was well-appearing and not in acute distress. Vital signs were stable: pulse rate 99 beats/min, respiratory rate 24 breaths/min, temperature 36.6 °C, and oxygen saturation 98 % on room air. Physical examination revealed a 2 × 2 cm firm, pulsatile, non-tender mass on the medial aspect of the left proximal arm, located 8 cm above the medial epicondyle. The overlying skin was intact with no discoloration. Left radial and ulnar pulses were palpable with normal volume. Sensory and motor functions of radial, ulnar and median nerves were intact. Lab tests showed Haemoglobin 13.6 g/dL, normal platelets, WBC, renal function electrolytes and ESR was within the normal range. Echocardiography was normal. Doppler ultrasound revealed a proximal brachial artery aneurysm with turbulent flow and normal distal radial/ulnar flow. Screening of major vascular beds showed no aortic or systemic aneurysms. CTA showed a well-defined round proximal brachial artery saccular dilatation measuring 1.7 × 1.6 cm, located over the anteromedial aspect of left arm, with no filling defect or calcification (Fig. 1a-c).

Fig. 1.

Preoperative CT angiography images of the left brachial artery aneurysm.

(a) Axial CT angiography demonstrating a well-defined saccular aneurysm (arrow) measuring 1.7 × 1.6 cm arising from the proximal left brachial artery without intraluminal thrombus.

(b) Sagittal reconstruction showing the relationship of the aneurysm to surrounding structures.

(c) Three-dimensional volume-rendered reconstruction illustrating the overall architecture of the aneurysm and its relationship to the brachial artery.

3. Surgical management

Under general anaesthesia, the patient was positioned supine with the left arm extended. After standard sterile preparation of the left upper extremity and left groin, a longitudinal skin incision was made over the site of the swelling (Fig. 2a). The brachial artery's proximal and distal segments were dissected and loop-controlled, with the aneurysm carefully isolated from adjacent neurovascular structures (Fig. 2b). A 5-cm segment of GSV was harvested from the left groin after careful dissection and branch ligation (Fig. 3a). The harvested vein was prepared by gentle distension with heparinized saline. Following systemic heparinization (1500 IU based on body weight), vascular control was achieved. The aneurysm sac was opened longitudinally, revealing a smooth internal wall without intraluminal thrombus (Fig. 2c). Reconstruction involved a reversed GSV graft with spatulated ends and interrupted 6-0 Prolene sutures for proximal and distal anastomoses (Fig. 3b). Heparinized saline was flushed before completing the anastomosis. After clamp release, excellent pulsatile flow was achieved with readily palpable distal radial and ulnar pulses. The aneurysm sac was partially excised, preserving surrounding structures and sent for histologic examination and the wound was closed in layers. Total operative time was 150 min with estimated blood loss of 70 mL.

Fig. 2.

Intraoperative findings of the brachial artery aneurysm.

(a) Preoperative skin marking showing planned longitudinal incision over the anteromedial aspect of left upper arm.

(b) Surgical exposure showing the brachial artery aneurysm after careful dissection. Note the proximal and distal healthy segments of brachial artery controlled with vessel loops. The aneurysm demonstrates typical outpouching with clear neck from the native vessel.

(c) Longitudinally opened aneurysm sac revealing smooth internal wall architecture without evidence of intraluminal thrombus or other changes, consistent with true aneurysm morphology.

Fig. 3.

Surgical reconstruction using reversed great saphenous vein graft.

(a) Harvested segment of great saphenous vein (GSV) measuring 5 cm in length from the left thigh, appropriately sized for interposition grafting.

(b) Completed reconstruction showing reversed GSV interposition with patent proximal and distal anastomoses. Note the partially excised aneurysm sac maintained to prevent injury to surrounding structures.

The patient had an uneventful recovery. Oral feeding began 8 h post-op, and pain was managed with Paracetamol suppositories. Peripheral pulses remained strong with normal motor and sensory function. Aspirin was started and continued for 30 days, and the patient was discharged on post-op day 2 without complications. Histopathology of the resected aneurysm wall revealed a true arterial aneurysm with focal medial disruption, neovascularization in the adventitia and mixed inflammatory infiltrate consisting of polymorphonuclear cells, plasma cells and lymphocytes suggesting acute on chronic non-specific inflammation (Fig. 4). At 3-month follow-up, graft patency and limb growth remain normal. Lifelong surveillance is planned.

Fig. 4.

Histopathological examination of the aneurysm wall.

(a) and (b). Histopathological examination (H&E stain, 100× magnifications) of the aneurysm wall showing all three layers of the arterial wall (intima, media, and adventitia) focal disruption and loss of smooth muscle cells in the tunica media and neovascularization in the adventitia. Mixed inflammatory infiltrate is present, composed predominantly of polymorphonuclear cells, plasma cells, and lymphocytes. Findings confirm the diagnosis of true arterial aneurysm with associated inflammatory changes.

4. Discussion

Idiopathic TBAA in children are extremely rare PAA, with few cases reported. This rarity poses significant challenges in establishing standardized management protocols, making each documented case valuable for expanding the collective clinical knowledge. These lesions may appear as painless masses or cause pain, ischemia or nerve compression. [[1], [2], [3], [4], [5], [6], [7]].

PAA in children are often discovered incidentally by parents, as in our case or during routine paediatric visits. Diagnosis typically relies on imaging such as ultrasonography, CTA or MRA [3,5,10]. Careful imaging selection and minimizing radiation is crucial for diagnosis, excluding other conditions and planning surgery in this age group [5]. Ultrasound, as shown in our case, is the preferred initial diagnostic tool. It also aids in screening, however it is less sensitive compared to CTA or MRA [5]. For unclear mass morphology, etiology or detailed operative planning, MRA or CTA may be required. Echocardiograms should also be used to rule out Kawasaki disease with coronary artery aneurysms [3,5]. In our case, diagnosis progressed systematically from ultrasound to CTA, for accurate surgical planning and ruling out other etiologies. While MRA is favoured for paediatric vascular imaging to minimize radiation, we chose CTA for its superior spatial resolution and detailed visualization of other vascular structures. This approach aligns with literature supporting CTA's diagnostic advantages in cases needing precise preoperative planning [[3], [4], [5]].

The Ann Arbor classification by Sarkar et al. [11] describes nine types of childhood aneurysms by etiology and pathogenesis. Class VIII covers rare congenital-idiopathic aneurysms, unrelated to associated disease, commonly involving the abdominal aorta, axillobrachial or iliofemoral vessels. These may present as pulsatile masses or embolic events with histology showing fibrous walls, medial thinning, elastic lamina fragmentation and secondary intimal fibroplasia [3,11]. The histopathological findings (Fig. 4) in our case aligns with this category, the presence of all three vessel wall layers confirms the diagnosis of a true aneurysm [1]. The observed focal disruption and loss of smooth muscle cells in the media layer, which has been reported in cases of congenital arterial aneurysms are often result of secondary changes. The presence of neovascularization in the adventitia, along with mixed inflammatory infiltrate, might indicate an active remodelling process rather than a primary anatomical defect [3,11].

Managing paediatric brachial artery aneurysms poses unique challenges. Reconstruction techniques depend on the aneurysm's size, location and nature, with considerations for growth and long-term vascular patency [[1], [2], [3], [4],7,10]. Some authors advise surgical reconstruction for all paediatric upper limb PAA's, regardless of size or symptoms [10]. While small, asymptomatic aneurysms in very young children may be monitored, moderate-to-large, enlarging or symptomatic aneurysms require early surgical intervention with favourable outcomes in most case series [[1], [2], [3], [4], [5], [6], [7],10]. Definitive surgical treatment involves aneurysm resection with primary end-to-end repair if feasible or reconstruction using a vein graft interposition or bypass grafting when tension is a concern. While synthetic grafts are an option, autologous veins are preferred in children for their growth potential, durability and lower infection risk and vein interposition grafting is often preferred with good outcomes reported in most case reports [2,4,6,7,11]. In our case we chose a GSV for reconstruction to avoid tension and because of its proven success in similar paediatric cases reported in the literature. Timely recognition and referral of paediatric vascular anomalies are vital to prevent complications particularly in resource limited settings. Given the potential for future aneurysms or related conditions, lifelong follow-up is recommended for these children for monitoring graft patency and limb growth [3,8].

5. Conclusion

This case demonstrates the successful surgical management of a rare paediatric isolated idiopathic TBAA with autologous vein graft reconstruction. While the optimal management strategy for these rare cases continues to evolve, this case contributes valuable insights to the limited literature on paediatric PAA and we emphasize the importance of early intervention and timely referral to a vascular specialist to prevent complications. Long-term follow-up and further studies are essential to understand growth adaptation and outcomes in paediatric vascular reconstructions.

Author contribution

Dagim Leykun Berhanu, MD: Conceptualization and design, writing original draft, literature review & editing and critical review of the paper.

Dereje Berhanu Mendere, MD: Literature review, writing the paper and editing and critical review of the paper.

Zerihun Gadisa chala, MD: Literature review, writing and drafting the paper, acquisition of data.

Fikir Awraris Bekele, M.D.: Literature review, writing and drafting the paper, acquisition of data.

Consent

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

Ethical approval

Ethical approval for this case report was exempted by Hawassa University College of Medicine and Health Sciences, as per institutional guidelines for case reports.

Guarantor

Dagim Leykun Berhanu, MD.

Research registration number

Not applicable.

Funding

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

Conflict of interest statement

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.