CASE REPORT |
https://doi.org/10.5005/jp-journals-10002-1472 |
Aberrant Right Subclavian Artery and Relevance in Neck Surgery: A Case Report
1,4Department of General Surgery, Yong Loo Lin School of Medicine, Singapore
2,3Department of Endocrine Surgery, National University Hospital, Singapore
Corresponding Author: Rajeev Parameswaran, Department of General Surgery, Yong Loo Lin School of Medicine, Singapore, Phone: + 65 67724229, e-mail: Chithram2025@icloud.com
Received: 13 October 2024; Accepted: 06 November 2024; Published on: 06 March 2025
ABSTRACT
Aberrant right subclavian artery (ARSA) is also known as ”lusorial artery” and is a developmental anomaly whereby the artery courses posterior to the esophagus as a branch of the arch of the aorta. The condition is commonly associated with a nonrecurrent laryngeal nerve (NRLN). Here, we report three cases of ARSA associated with NRLN during thyroid and parathyroid surgery, detected during routine computerized tomography of the thorax. Patients commonly present with dysphagia, which can be disabling at times. In patients with severe symptoms associated with ARSA, vascular interventions may be required with open, thoracic endovascular aortic repair (TEVAR), or hybrid procedures.
Keywords: Case report, Subclavian artery, Thyroid parathyroid
How to cite this article: Soo J, Tan L, Pinto D, et al. Aberrant Right Subclavian Artery and Relevance in Neck Surgery: A Case Report. World J Endoc Surg 2024;16(2):48–52.
Source of support: Nil
Conflict of interest: Dr Rajeev Parameswaran is associated as the Associate Editorial Board member of this journal and this manuscript was subjected to this journal’s standard review procedures, with this peer review handled independently of this editorial board member and his research group.
Patient consent statement: The author(s) have obtained written informed consent from the patient(s) for publication of the case report details and related images.
INTRODUCTION
Aberrant right subclavian artery (ARSA) is an anatomical variant where the right subclavian artery, which normally arises from the brachiocephalic artery, originates from the aortic arch. Though ARSA is the most common abnormality originating from the aortic arch, its occurrence is rare, ranging from 0.2 to 1.7%.1,3 Previously, an ARSA classification system considering the branching variations was suggested and proposed as an aid to descriptive communication and surgical planning, but this has not been widely adopted.4
Most patients with ARSA are asymptomatic, but its clinical importance arises during procedures involving regional anatomy. Multiple case series have reported ARSA found during esophagectomy,5,6 parathyroidectomy, and thyroidectomy.7,9 Considering the domain of thyroid and parathyroid surgery, ARSA is commonly associated with the occurrence of nonrecurrent laryngeal nerve (NRLN),10,11 where potential iatrogenic injury is the most feared complication.12,13
This article describes our experience with three patients with ARSA and the importance of the presence of ARSA in thyroid and parathyroid surgery.
METHODS
The study is a retrospective review of 3,339 patients who underwent thyroidectomy from 2000 to 2021 at a single tertiary institution. Patients who had an NRLN and ARSA were identified, and their clinicopathological details were collected. Over the time period, there were three patients with an aberrant subclavian artery detected on computerized tomography, and the details pertaining to the three cases are described below. A brief review of the literature using PubMed with the keywords ”aberrant,” ”subclavian artery,” ”thyroid,” ”parathyroid,” and ”surgery” was also performed for articles published in English between 2000 and 2022.
Case 1
A 72-year-old female was referred to the clinic with progressive compressive symptoms of dysphagia and dysphonia over the course of a year. She underwent a subtotal thyroidectomy in 1985, followed by a redo thyroidectomy in 2010, and in both surgeries, her RLN was reported to be in normal anatomical position. In view of her third recurrence and the presence of a large goiter, a CT scan (Fig. 1) was performed, which showed the presence of ARSA, type II, raising the suspicion of an NRLN. In 2016, the patient underwent a revision thyroidectomy during which a type I NRLN was identified. She sustained a nerve palsy from which she recovered and continues to be well with no symptoms.
Figs 1A and B: CT scan of neck and thorax from patient 1 who presented with large recurrent symptomatic goiter. (A) CT neck showing the large recurrent goiter with tracheal compression and deviation; (B) Retrotracheal ARSA (shown by pointed red arrow)
Case 2
A 62-year-old female was referred to the endocrine surgical clinic with symptoms of severe dysphagia and a nodular goiter, predominantly on the right side. The patient’s past medical history included repair of an ASD in childhood, asthma, and gastroesophageal reflux disease (GERD). She underwent an uncomplicated hemithyroidectomy in 2012, and the operative notes recorded the RLN in normal anatomical position. The symptoms of dysphagia persisted, for which she underwent evaluations with upper gastrointestinal endoscopy, which showed mild reflux disease. A barium swallow performed showed indentation of the posterior esophagus by extrinsic compression, and a CT scan showed the presence of a type III ARSA (Fig. 2). In view of the lusorial artery causing severe dysphagia, reimplantation of the ARSA was recommended, but the patient declined to undergo the surgery.
Figs 2A and B: Dysphagia lusoria with type I ARSA. (A) Barium swallow from patient 2 showing the indentation of the esophagus by ARSA (shown by yellow pointed arrow); (B) ARSA running posterior to the trachea and esophagus (shown by red pointed arrow)
Case 3
A 58-year-old man was referred for consideration of total parathyroidectomy for renal hyperparathyroidism, with a background history of end-stage renal failure (ESRF) from lupus nephritis and bilateral nephrectomy for renal carcinoma in 2014. He had been undergoing hemodialysis since 2014 and was also undergoing treatment for diabetes and hypertension. A CT scan was performed as part of the evaluation for central venous stenosis, which showed the presence of an ARSA. At surgery, using intraoperative neuromonitoring (IONM), a type II NRLN was identified (Fig. 3). His surgery was uncomplicated, and he was discharged back to the care of the renal physicians.
Figs 3A and B: ARSA as seen in patient 3 who underwent surgery for renal parathyroidectomy. (A) Intraoperative finding of right NRLN (type II) (highlighted by yellow star) coursing under the ITA; (B) Retrotracheal ARSA arising from the aortic trunk
DISCUSSION
Normally, the aortic arch has three branches, namely the brachiocephalic trunk, left common carotid, and subclavian artery in this order from right to left. The brachiocephalic trunk, in turn, gives rise to the right common carotid artery and subclavian artery. During normal embryological development, fusion of the right seventh intersegmental artery takes place with the right dorsal aorta.3 Failure of fusion of the two arteries results in the formation of ARSA, leading to an abnormal involution of the proximal right dorsal aorta and a persistent right seventh intersegmental artery formed from the proximal descending thoracic aorta.2 The frequency of ARSA ranges between 0.2 and 13% in the literature, and it is more commonly seen in women.14,16 The condition is commonly associated with atrial septal defects, as seen in the case of patient 2, Down’s syndrome, with a prevalence of about 24%.17 It is less commonly seen in DiGeorge syndrome, associated with chromosome 22q11 deletion (CATCH 22).18 ARSA can also be seen with a right-sided aortic arch on the left side, seen in 0.1% of the population.19 It can also be associated with Kommerell’s diverticulum (aneurysmal dilatation of the aorta).20,21
Hunauld in 1735 first described ARSA, and in 1794, David Bayford reported on the association of a form of dysphagia with the presence of ARSA, which he termed dysphagia lusoria.22 The term ”lusoria” artery has been loosely applied to ARSA, mainly in patients with symptomatic dysphagia. The course that the ARSA takes from the arch is of three types (Fig. 4): type I, where the artery courses posterior to the esophagus; type II, between the esophagus and the trachea; and type III, where the artery courses anterior to the trachea. The most common abnormality seen is type I (~80%), type II (~15%), and type III (~5%).23 With the retroesophageal and retrotracheal course of ARSA, patients can present with symptoms of dysphagia, thoracic pain, dyspnea, sometimes persistent cough, and unexplained ischemia of the right upper limb.24 Further complications such as hematemesis or hemoptysis can occur due to arterio-esophageal or arterio-tracheal fistula, respectively, and possibly rupture of the aneurysm.25,26 However, most patients with ARSA usually have no symptoms, and the anatomical variant is found incidentally during imaging of the thorax, surgical procedures involving its regional anatomy, or during autopsies.
Fig. 4: Types of ARSA course in relation to the trachea and esophagus. Type I (most common type)—ARSA courses posterior to the trachea and esophagus. Type II—ARSA courses between the trachea and esophagus. Type III—ARSA lies in front of trachea and esophagus. Abbreviations: ARSA, aberrant right subclavian artery; BT, brachiocephalic trunk; CCA, common carotid artery; SA, subclavian artery
Relevant to thyroid and parathyroid surgery, the presence of ARSA results in the variation of the course of the RLN called NRLN.10,12 NRLN described in the literature are of three types: type I runs closely to the superior thyroid pedicle; type II runs in parallel to the inferior thyroid artery (ITA) and transversely above the artery; and type III runs parallel to the ITA and transversely between or under the ITA.27 Previous studies that reported on NRLN in relation to ARSA shared variations to the aforementioned three types: one cited that the course of the vagus nerve in the carotid sheath ran more medially than usual,28 and another cited that the branching level of the NRLN could be unpredictable, as observed in their case series in positions such as at the cricoid cartilage or at the inferior pole of the thyroid.29 These observations must be kept in mind by surgeons when embarking on thyroid and parathyroid surgery, and certain pointers have been described to help identify the presence of NRLN in neck surgery.12
Preoperative assessment should take place with emphasis on airway, as cases requiring prolonged endotracheal intubation can trigger gastrointestinal bleeding in patients with fistula.30 Counseling of risks can include the potential risk of tracheostomy bleeding if patients are known to have ASCA preoperatively, with necessary communication related to risks of bilateral recurrent laryngeal nerve injury and resultant cord palsy requiring tracheostomy.31 In patients with severe symptomatic disease, surgical intervention is necessary, and few techniques have been reported. There is no level 1 evidence to recommend interventions for ARSA, given the rarity of the condition, but the consensus is that ARSA with a diameter of 3 cm or more needs intervention due to the risk of dissection or rupture.32 Where patients require intervention, this may be managed with open repair,21 thoracic endovascular aortic repair (TEVAR),33,34 or hybrid repair, and the choice of procedures depends on factors such as age of the patient, symptoms, morphology of the vessels, and acuity of presentation.32 Hybrid or TEVAR repair is preferred due to the high risk of morbidity and mortality associated with open repair, which is close to 16%.21,32
CONCLUSION
Failure to recognize the presence of ASCA has implications on associated anatomy and surgical outcomes. Knowledge of this anatomical variation is important to thyroid surgeons to minimize operative complications and its impact on the patient’s quality of life.
ORCID
Rajeev Parameswaran https://orcid.org/0000-0002-3318-0357
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