ANTICANCER RESEARCH 33: 1635-1640 (2013)

Successful Resection of with Right Aortic Arch by Video-Assisted Thoracoscopic Surgery: A Case Report

NAOSHI KUBO1, MASAICHI OHIRA1, TOMOHIRO LEE1, KATSUNOBU SAKURAI1, TAKAHIRO TOYOKAWA1, HIROAKI TANAKA1, KAZUYA MUGURUMA1, KENJIRO KIMURA1, HISASHI NAGAHARA1, EIJI NODA1, RYOSUKE AMANO1, HIROSHI OHTANI1, YOSHITO YAMASHITA2, MASAKAZU YASHIRO1, KIYOSHI MAEDA1 and KOSEI HIRAKAWA1

1Department of Surgical Oncology, Osaka City University Graduate School of Medicine, Osaka, Japan; 2Department of Gastroenterological Surgery, Osaka City General Hospital, Osaka, Japan

Abstract. The right aortic arch (RAA) forms a vascular to understand the anatomy of these systems in order to ring, encircling both the and trachea. We herein safely perform an operation. Recently, video-assisted report a case of thoracic esophageal cancer with RAA thoracoscopic surgery (VATS) has been applied in successfully resected using video-assited thoracoscopic esophagectomy and has been reported to deliver better surgery (VATS). A 64-year-old man who presented with a surgical outcomes. VATS for patients with esophageal complaint of abdominal pain, was admitted to our hospital. cancer has several advantages, such as a good surgical view Further examinations revealed gall stones and multiple by means of visual enlargement, and less trauma to the superficial esophageal carcinomas. Three-dimensional thoracic wall, compared with conventional open computed tomographic images showed RAA and aortic thoracotomy. We herein report a case of thoracic diverticulum. The trachea and esophagus were completely esophageal cancer with RAA, successfully resected using encircled by the RAA, the aortic diverticulum and the VATS. All previously reported cases with esophageal cancer pulmonary artery. We successfully and safely performed associated with RAA underwent open esophagectomy. To VATS esophagectomy for esophageal cancer accompanied the best of our knowledge, this case is the first report of a with RAA. To the best of our knowledge, this report is the patient undergoing VATS esophagectomy for esophageal first case for which VATS esophagectomy for esophageal cancer associated with RAA. We were easily able to cancer, accompanied with RAA, was carried out. Marking of understand the unusual anatomical localization of RAA and the left recurrent laryngeal nerve with colored tape at the safely dissected the mediastinal lymph nodes by visual cervical part of the surgery made it easy to identify it during enlargement thorough the thoracoscope. In addition, it was the thoracoscopic part of the surgery. very useful to mark the left recurrent laryngeal nerve (LRLN) with colored tape during the cervival portion of the Right-sided aortic arch (RAA), which is a rare vascular surgery before the thoracoscopic portion of the surgery malformation, forms a vascular ring encircling both the started. esophagus and trachea. Consequently, esophagectomy for esophageal cancer associated with RAA is extremely rare; Case Report only approximately 30 such cases have been reported. Because most cases with RAA have vessel and nervous A 64-year-old man visited a local physician with a complaint system anomalies in the mediastinum, it is very important of abdominal pain. Further examinations showed acute cholecystitis due to gallstones. Esophageal cancer was accidentally found by upper gastrointestinal endoscopic examination. The patient Correspondence to: Naoshi Kubo, Department of Surgical was admitted to our hospital for surgical treatment for accute Oncology, Osaka City University Graduate School of Medicine, 1- cholecystitis and esophageal cancer. 4-3 Asahi-machi, Abeno-ku, 545-8585, Osaka Japan. Tel: +81 Chest radiography demonstrated an RAA with normal 666453838, Fax: +81 666466450, e-mail: [email protected] cardiac findings. cu.ac.jp Barium esophagography showed right indentations in the Key Words: Right aortic arch, esophageal cancer, video-assisted upper thoracic esophagus at the point of the aortic arch, and thoracoscopic surgery (VATS). no tumor was identified (Figure 1A). Esophagoscopy showed

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Figure 1. A: Barium esophagography showing the indentation of the upper thoracic esophagus at the point of a right aortic arch (arrowhead). B: Esophagoscopy with the iodine stain showing multiple depressed lesions located at the middle and lower thoracic esophagus.

multiple superficial depressed lesions located approximately route in the supine position. The second step was subtotal 30-32, 35-37, and 38-39 cm distal from the incisors (Figure esophagectomy and mediastinal lymph node dissection using 1B). Biopsy samples of the lesions revealed moderately- VATS in the right lateral position. For the first step, the differentiated squamous cell carcinomas. Three-dimensional patient was positioned in the supine position. Through a computed tomographic (3D-CT) images showed an RAA and cervical collar incision, lymphadenectomy along the left and aortic diverticulum, known as Kommerell’s diverticulum right recurrent laryngeal nerve and above both clavicles was (Figure 2). The trachea and esophagus were completely performed by a cervical team. Subsequently, the LRLN was encircled by the RAA, Kommerell’s diverticulum and the marked with colored vessel tape. Simultaneously, an pulmonary artery. Chest 3D-CT revealed that the right abdominal team performed and mobilized common carotid artery and subclavian artery branched from the by hand-assisted laparoscopic surgery. the RAA, while the left common carotid artery and subclavian Subsequently, celiac and perigastric lymph node dissection artery branched from Kommerell’s diverticulum. From these was performed, after which a 4-cm width gastric conduit was findings, we diagnosed multiple superficial esophageal made extracorporeally by an automatic suturing instrument. carcinomas accompanied with an RAA and gallstones. The gastric conduit was pulled up through a retrosternal Subtotal esophagectomy and three-field lymphadenectomy route and cervical esophago-gastric anastomosis was using VATS and cholecystectomy was performed. performed. The patient was then turned into the right lateral position and underwent a subtotal esophagectomy and Operative findings. The operation consisted of two steps. The mediastinal lymphadenectomy with left minithoracotomy first step was cervical and abdominal lymphadenectomy and under VATS. A 5-cm minithoracotomy was made in the reconstruction using a gastric tube through the retrosternal fourth intercostal space (ICS) on the anterior axillary line,

1636 Kubo et al: Resection of Esophageal Cancer with RAA by VATS

Figure 2. Three-dimensional computed tomography (3D-CT): anteroposterior view (A), left lateral view (B). 3D-CT showed the right aortic arch (RAA) accompanied by aortic diverticulum (AD) known as Kommerell’s diverticulum. The left common carotid artery (LCA) and left subclavian artery (LSA) arose from the AD. RSA: Right subclavian artery, RCA: right common carotid artery.

where the camera and metal retractor were inserted. Three Discussion other trocars were inserted as follows: One 10-mm trocar for an assistant was placed at the second ICS on the anterior RAA is a rare vascular anomaly and has been reported to be axillary line, and two 10-mm trocars for the operator were present in 0.04-0.1% of autopsy cases in Western countries placed at the fourth ICS on the posterior axillary line and (2). Because the trachea and esophagus of a patient with fifth ICS on the middle axillary line. The thoracoscope RAA are compressed by a vascular circle, most patients revealed that the trachea and esophagus were completely with this malformation have respiratory symptoms such as encircled by a vascular ring consisting of an aortic arch, stridor or gastrointestinal symptoms, choking with food aortic diverticulum, left ductus arteriosus and pulmonary from infancy and early childhood (3). Edwards et al. (4) artery. The left ductus arteriosus was found to be closed and identified three types of aortic anomalies based on a was preserved. The LRLN marked with colored vessel tape theoretical concept of the development of the aortic arch. at the cervical procedure was easily identified and preserved. RAA is Group III of this classification. Stewart et al. also The LRLN passed behind the left ductus arteriosus and showed RAA was in turn divided into three subtypes (5) ascended posteriorly (Figure 3). Subtotal esophagectomy (Figure 4): subtype I exhibits a mirror image branching of with radical dissection of para-esophageal and mediastinal the normal left aortic arch, subtype II an aberrant left lymph nodes was performed. Lymph nodes along the right subclavian artery, and subtype III a left subclavian artery recurrent laryngeal nerve (RRLN) were dissected through a which is no longer connected. The present case incision. The postoperative course was uneventful and demonstrated the type of RAA with aortic diverticulum, the patient was discharged from the hospital 21 days while the left carotid artery and subclavian artery branched postoperatively. Macroscopic findings showed multiple from the aortic diverticulum (Figure 5). This case did not superficial esophageal cancers, and microscopic findings belong to any of the subtypes from the Stewart et al.’s showed moderately differentiated classification. The left ductus arteriosus connected the within the mucosal layer and no metastatic lymph nodes at aortic diverticulum and the pulmonary artery. The LRLN any site. According to the TNM classiffication version 7 (1), passed behind the left ductus arteriosus and ascended these tumors were classified as T1N0M0 stage IA. posteriorly. Shimakawa et al. (6) reviewed the as then 28

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Figure 3. The thoracoscopic view and schematic illustration of the upper mediastinum after esophagectomy with lymphadenectomy. The left recurrent laryngeal nerve (LRLN) arose from the vagus nerve (VN). The LRLN, marked with colored vessel tape, circled beneath the left ductus arteriosus (LDA) and ascended to the neck. AD: Aortic diverticulum.

reported cases of esophageal cancer with RAA and showed Lymph nodes around both recurrent laryngeal nerves are eight cases had RAA of subtype IIIA, while 19 cases had common sites of . Kinoshita et al. (7) stated that RAA of subtype IIIB according to Stewart et al.’s a median sternotomy should be added to a left thoracotomy classification. However, there was no report of esophageal in cases with advanced thoracic esophageal cancer with cancer with RAA, as found in the present case. Shimakawa RAA to resect completely the lymph nodes along the et al. reported that subtotal esophagectomy and mediastinal RRLN. Moreover, Noguchi et al. (8) reported that the left lymph-node dissection through a left thoracotomy have been door open method was useful for radical dissection of lymph performed in most cases of RAA with esophageal cancer. nodes along RRLN and LRLN. Since in the present case, All patients with esophageal cancer accompanied by RAA, the tumor depth was considered to be superficial and the underwent open esophagectomy through a left thoracotomy. preoperative chest CT and PET-CT showed no evidence of Left thoracotomy has been reported to give a better view lymph node metastasis around the RRLN, we performed the into the mediastinum for esophagectomy in patients with lymph node dissection along the RRLN through the neck RAA than an approach through the right thoracotomy incision alone, considering that sternotomy or left door open because RAA and right-sided descending aorta interfere method would add unnecessary stress. with the mobilization of the esophagus located at the left One of the most important parts of thoracic side of the RAA through a right thoracotomy. However, it esophagectomy for esophageal cancer is the LN dissection is difficult to carry out an adequate dissection of lymph along the LRLN. Shimakawa et al. Reported that in most nodes around the RRLN through a left thoracotomy alone. cases with RAA, the LRLN branched from the vagal nerve

1638 Kubo et al: Resection of Esophageal Cancer with RAA by VATS

Figure 4. Classification of the right aortic arch by Stewart et al. (4): Right aortic arch with mirror-image branching (type I), with aberrant left subclavian artery (LSA) (type II), with isolation of the left LSA (type III). RSA: Right subclavian artery, RCA: right common carotid artery, LCA: left common carotid artery.

and passed behind the left ductus arteriosus and ascended to the neck along the trachea. We were easily able to identify the LRLN, as it had been marked with colored vessel tape at the first-step cervical procedure. This procedure was very useful in this case because RAA had unusual vessel anomalies and nerve system. VATS for esophageal cancer was first reported by Cuschieri et al. (9) in 1992. Since then, many reports of the advantages and non-inferiority of VATS compared to open surgery for esophageal cancer have been published (10, 11). At our institute, VATS esophagectomy was first applied for esophageal cancer in July 2000 and since then has been performed safely for about 140 patients with esophageal cancer at stages up to T3 with regional lymph node metastasis without severe pleural adhesion. In this case, we were easily able to understand the unusual anatomical situation of RAA and safely dissected the mediastinal lymph nodes by visual enlargement thorough the thoracoscope. To our knowledge there is no report of esophageal cancer accompannied with RAA treated with VATS esophagectomy. In summary, we successfully performed the first reported VATS esophagectomy for esophageal cancer accompanied by Figure 5. A schematic illustration of the mediastinal structure of the RAA. We recommend VATS esophagectomy by a well- present case. The trachea and esophagus were encircled completely by trained team of endoscopic surgeons for patients with the right aortic arch (RAA), aortic diverticulum (AD) and left ductus esophageal cancer with RAA. In addition, marking of the arteriosus (LDA). The AD gave rise to the left subclavian artery (LSA) and left common carotid artery (LCA). RSA: Right subclavian artery, LRLN with vessel tape at the cervical part of the surgery RCA: right common carotid artery, LCA: left common carotid artery, made it easy to identify it during the thoracoscopic part of DA: descending aorta, PA: pulmonary artery, VN: vagus nerve, LRLN: the surgery. left recurrent laryngeal nerve, T: trachea, E: esophagus.

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