Eur J Cardiothorac Surg 2002;22:64-69
© 2002 Elsevier Science NL
Resection of Kommerell's diverticulum and left subclavian artery transfer for recurrent symptoms after vascular ring division
C.L. Backera,b*,
N. Hillmana,b,
C. Mavroudisa,b,
L.D. Holingera,c
a Department of Surgery, Northwestern University Medical School, Chicago, IL, USA
b Division of Cardiovascular-Thoracic Surgery, Children's Memorial Hospital, Chicago, IL, USA
c Division of Pediatric Otolaryngology, Children's Memorial Hospital, Chicago, IL, USA
Received 13 September 2001;
received in revised form 29 January 2002;
accepted 26 March 2002.
* Corresponding author. Division of Cardiovascular-Thoracic Surgery, M/C #22, Children's Memorial Hospital, 2300 Children's Plaza, Chicago, IL 60614, USA. Tel.: +1-773-880-4378; fax: +1-773-880-3054
e-mail: c-backer{at}northwestern.edu
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Abstract
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Objectives: A Kommerell's diverticulum in patients with a right aortic arch may become aneurysmal and be an independent cause of tracheoesophageal compression, even after ligation and division of a left ligamentum. We review the indications for and results of Kommerell's diverticulum resection and left subclavian artery transfer in children with a right aortic arch who previously underwent vascular ring (ligamentum) division. Methods: From 1998 through 2001, eight children have been referred with recurrent respiratory symptoms (n=8) and/or recurrent dysphagia (n=4) after vascular ring division. Each child had a right aortic arch with a left ligamentum and had undergone division of the ligamentum elsewhere. All had a Kommerell's diverticulum that was not addressed at the initial operation. All patients had a repeat left thoracotomy with resection of the diverticulum. Five patients had division and reimplantation of the left subclavian artery into the left carotid artery to relieve the sling-like effect of the retroesophageal left subclavian artery on the right aortic arch. One other patient had primary Kommerell's diverticulum resection and transfer of the left subclavian artery to the left carotid artery. Results: The mean age at the initial operation was 1.7±0.9 years, and the mean age at reoperation was 8.0±3.7 years. In all patients postoperative bronchoscopy confirmed relief of the tracheal compression. There were no complications related to the subclavian artery transfer. Two patients developed postoperative chylothorax, one requiring thoracic duct ligation. The median hospital stay was 5 days. All patients had dramatic resolution of their preoperative symptoms. Conclusions: Kommerell's diverticulum is an important anatomic structure that can cause recurrent symptoms in patients with a right aortic arch after ligamentum division. In selected patients, reoperation with resection of the Kommerell's diverticulum and transfer of a retroesophageal left subclavian artery results in relief of symptoms. This technique has become our procedure of choice as a primary operation for children with a right aortic arch and a significant Kommerell's diverticulum.
Key Words: Vascular ring • Right aortic arch • Kommerell's diverticulum
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1. Introduction
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‘Kommerell's diverticulum’ was first described by B. Kommerell [1] in a German radiology journal in 1936. He described a patient with a left aortic arch and an aberrant right subclavian artery arising from a ‘diverticulum of the aorta’ from the descending thoracic aorta. He postulated that the embryologic origin of this diverticulum was the dorsal right fourth aortic arch. In patients with a right aortic arch, this same diverticulum can be the source of the left subclavian artery arising from the descending thoracic aorta (Fig. 1)
. In this case the diverticulum is an embryologic remnant of the dorsal left aortic arch [2]. Kommerell's diverticulum has been known to compress the trachea and esophagus independent of the action of the right aortic arch and left ligamentum in children with vascular rings. Several groups [3–6] have previously reported patients with a right aortic arch who had recurrent symptoms after vascular ring division (ligamentum division) who then had subsequent successful excision of a Kommerell's diverticulum with complete relief of symptoms. Aneurysm formation of a Kommerell's diverticulum in a patient with a left aortic arch and aberrant right subclavian artery with successful surgical resection has also been described [2,7,8].
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Fig. 1. This illustration shows the typical anatomy of a patient with a right aortic arch, retroesophageal left subclavian artery, and large Kommerell's diverticulum. The Kommerell's diverticulum is an embryologic remnant of the left fourth aortic arch. LCA/RCA, left/right carotid artery; LSA/RSA, left/right subclavian artery.
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The purpose of this review is to summarize the clinical presentation, diagnostic evaluation, surgical techniques, postoperative complications, and long-term outcome in patients with a right aortic arch and recurrent symptoms secondary to a Kommerell's diverticulum after ligamentum division.
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2. Patients and methods
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This series has as a background an extensive historical experience with vascular rings at Children's Memorial Hospital. Between 1947 and 2001, this includes treatment of over 340 patients with various types of vascular rings, as shown in Table 1. From 1998 through 2001, eight children have been referred with recurrent respiratory symptoms (n=8) and/or recurrent dysphagia (n=4) after vascular ring division. Respiratory symptoms included chronic cough, wheezing, recurrent upper respiratory tract infections, recurrent pneumonia, and exercise intolerance. Each child had a right aortic arch with left ligamentum and had undergone division of the left ligamentum through a left thoracotomy elsewhere. Preoperatively, all patients had anteroposterior and lateral chest radiographs, all patients had an esophagogram, and all patients had a CT or MRI of the thorax. Several patients had angiograms performed. All patients had preoperative bronchoscopy. In all patients there was a Kommerell's diverticulum originating from the descending aorta near the site of the previous ligamentum division. The subclavian artery originated from the Kommerell's diverticulum in six patients. Six patients had a retroesophageal left subclavian artery, and two had an anterior origin of their left subclavian artery (mirror-image branching) (Fig. 2)
[9]. All had a Kommerell's diverticulum that was not addressed at the initial operation.
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Fig. 2. Cephalic view of the two most common types of right aortic arch; right aortic arch with mirror-image branching and right aortic arch with retroesophageal left subclavian artery. In both cases shown here there is a Kommerell's diverticulum and in both cases the ligamentum arteriosum completes the vascular ring. AAo, DAo, ascending, descending aorta; LCA, RCA, left, right carotid artery; LPA, MPA, left, main pulmonary artery; LSA, RSA, left, right subclavian artery.
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All patients had a repeat left thoracotomy with resection of the diverticulum (Fig. 3)
. All patients had lysis of adhesions from the prior surgery. Five patients had division and reimplantation of the left subclavian artery into the left carotid artery (Fig. 4) . This anastomosis was done with 7-0 running polypropylene sutures. The transfer was performed to relieve the sling-like effect of the retroesophageal left subclavian artery on the right aortic arch (Fig. 5)
. In seven patients the diverticulum was resected flush with the aorta, and in one a Gore-Tex patch (polytetrafluoroethylene, W.L. Gore & Associates, Flagstaff, AZ) was used to patch the resected area because the base of the diverticulum was rather friable. The patients with subclavian artery transfer have been kept on aspirin for 6 months postoperatively.
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Fig. 3. A schematic illustration of the resection of a Kommerell's diverticulum through a left thoracotomy. There is a vascular clamp partially occluding the descending thoracic aorta at the origin of the Kommerell's diverticulum. The Kommerell's diverticulum has been completely resected.
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Fig. 4. The completed repair. The orifice where the Kommerell's diverticulum was resected is usually closed primarily or, as shown in the inset, the orifice can be patched with polytetrafluoroethylene if necessary. The left subclavian artery has been implanted into the side of the left common carotid artery with fine running polypropylene suture.
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Fig. 5. Cephalic view of the result of ligamentum division, Kommerell's diverticulum resection, and left subclavian artery transfer to the left carotid artery. The panel on the left shows the compressed trachea and esophagus preoperatively. The panel on the right shows that the trachea and esophagus are no longer compressed postoperatively. Division of the ligamentum alone does not release the sling-like effect of the left subclavian artery on the right arch. AAo, DAo, ascending, descending aorta; LCA, RCA, left, right carotid artery; LPA, MPA, left, main pulmonary artery; LSA, RSA, left, right subclavian artery.
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A summary of the clinical characteristics of the children having Kommerell's diverticulum resection after prior vascular ring surgery is shown in Table 2. The mean age at initial operation was 1.7±0.9 years, with a range from 0.3 to 3 years. The mean age at the reoperation for Kommerell's diverticulum was 8.0±3.7 years, ranging from 2.5 to 13 years. The time interval between the two operations ranged from 1.5 to 12.7 years with a mean of 6.3±3.9 years. The postoperative hospital stay ranged from 3 to 23 days, with an average stay of 8.2±7.2 days and a median stay of 5 days. The median diverticulum size was 1.5x2 cm.
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Table 2. Clinical characteristics of patients having Kommerell's diverticulum resection after prior vascular ring surgerya
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A final patient, who did not have previous ligamentum division, recently underwent elective primary operation for a vascular ring. This 2.5-year-old child had a right aortic arch, retroesophageal left subclavian artery, left ligamentum, and a large Kommerell's diverticulum. This was demonstrated by preoperative barium swallow and MR imaging. Because of the large Kommerell's diverticulum, we elected to resect the Kommerell's diverticulum and implant the left subclavian artery into the left carotid artery primarily. Preoperative bronchoscopy demonstrated significant compression of the right trachea from the right arch and the posterior trachea from the Kommerell's diverticulum. Follow-up bronchoscopy showed complete resolution of the posterior compression of the trachea and significant improvement in the right-sided compression. She was discharged from the hospital 3 days postoperatively and at short follow-up remains asymptomatic. This was the first patient in our series of vascular ring patients where implantation of the left subclavian artery was performed into the left carotid artery as a primary operation.
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3. Results
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There was no operative or late mortality. No patient required a blood transfusion. In all patients postoperative bronchoscopy confirmed relief of the posterior tracheal compression from the Kommerell's diverticulum. No patient had a recurrent laryngeal nerve injury. No patient had a complication related to the subclavian artery transfer. Two patients (the first two in the series) developed a postoperative chylothorax. One patient required right thoracotomy and ligation of the thoracic duct. The other patient with chylothorax had resolution with treatment with a fat-free diet and intravenous hyperalimentation. The median hospital stay was 5 days. All patients have had dramatic resolution of their preoperative respiratory and swallowing symptoms. Our experience with the first seven patients led us to offer this as a primary operation to a recent patient with excellent results.
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4. Discussion
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Right aortic arch with left ligamentum to the descending aorta is one of the two true vascular rings that cause tracheoesophageal compression in children [10]. These children usually present somewhat later in life than children with a double aortic arch with symptoms of stridor, recurrent respiratory tract infections, recurrent pneumonia, barky cough, and often with dysphagia for solid foods in later years. In the series of patients operated on for vascular rings at Children's Memorial Hospital between 1947 and 2001, 90 of 344 patients (26%) have had a right aortic arch with left ligamentum. In our series, 65% of these patients had a retroesophageal left subclavian artery; in 35% there was mirror-image branching [11]. The classic therapy for this vascular ring has been left thoracotomy with division of the ligamentum [10,11]. Technical considerations that ensure the success of the procedure include division not only of the ligamentum but any fibrous bands crossing the esophagus in that area. In addition, the pleura that is opened to expose the aortic arch should be left open at the time of the procedure; two of the eight patients who underwent reoperation in this series had their pleura closed at the time of vascular ring surgery. This tends to cause recurrent scar tissue and seemed, in particular, to be associated with adhesive bands crossing the esophagus and was probably a significant part of the patients' postoperative dysphagia.
A small number of patients with a right aortic arch will have origin of the left subclavian artery from a Kommerell's diverticulum. This enlargement of the takeoff of the subclavian artery was originally described in a patient with a left aortic arch and aberrant origin of the right subclavian artery from the descending aorta [1]. A Kommerell's diverticulum in patients with a right aortic arch is an embryologic remnant of the left fourth aortic arch posteriorly. A Kommerell's diverticulum can enlarge and independently compress the trachea. In our series of patients with a right arch operated on primarily we have had three patients with a large Kommerell's diverticulum that was resected at the time of the initial vascular ring surgery. We now have in addition a recent patient in whom we performed elective resection of the Kommerell's diverticulum and reimplantation of the left subclavian artery into the left carotid artery at the time of vascular ring repair. The eight patients described in this current review all had recurrent symptoms from a Kommerell's diverticulum after simple ligamentum division. All were referred from other institutions, four from out of state. All patients had relief of symptoms after reoperation with Kommerell's diverticulum resection and transfer of the left subclavian artery occurred in five of the seven patients.
In the first patient in our series, the Kommerell's diverticulum was resected without transferring the left subclavian artery. In retrospect, we feel that this transfer probably should be performed in all patients with a retroesophageal left subclavian artery. The two factors that create tracheal compression in these patients are the space-occupying effect of the Kommerell's diverticulum and the sling-like effect of the left subclavian artery pulling the right aortic arch towards the left side. The resection of the Kommerell's diverticulum is intuitively obvious as a source of relief for these patients. The sling-like effect of the left subclavian artery is not as intuitively obvious, but the release of the right aortic arch, allowing it to fall into the right chest and away from the trachea, is fairly dramatic when pre- and postoperative bronchoscopy is performed on these patients. This result is schematically shown in Fig. 5.
The chief complication in this series has been chylothorax, which occurred in the first two patients in the series. One patient required thoracic duct ligation, while the other resolved with hyperalimentation. Since then, we have been much more aware of the potential for thoracic duct injury during the dissection. The dissection plane is best established anteriorly as the thoracic duct tends to be posterior. The dissection plane should then be kept close to the Kommerell's diverticulum as that helps avoid entering the thoracic duct. The first patient in the series developed a right chylothorax that was probably secondary to an actual occlusion of the thoracic duct during the dissection for the Kommerell's diverticulum resection. The occlusion of the thoracic duct in the left chest then led to a weeping of chyle from the right pleural surfaces. This resolved with ligation of the thoracic duct at the diaphragm through a right thoracotomy.
Jung et al. [3] reported five cases of right aortic arch with aberrant left subclavian artery and left ligamentum arteriosum. Three patients had a significant Kommerell's diverticulum. Two patients had as an initial procedure ligation and division of a ligamentum arteriosum through a left thoracotomy. Both patients required a second operation through a right thoracotomy; in one patient an aberrant left subclavian artery was ligated and divided, and in a second patient the aberrant left subclavian artery was ligated and divided and a Kommerell's diverticulum was excised. They also reported two patients who had primary ligation and division of an aberrant left subclavian artery and Kommerell's diverticulum resection through a right thoracotomy. They speculated that the potential hazard of left hand ischemia secondary to subclavian artery ligation or late subclavian steal syndrome could be prevented by reanastomosing the divided left subclavian artery into the left common carotid artery or the aorta. They noted that this could be accomplished through a left thoracotomy that would allow division of the ligamentum and reimplantation of the aberrant left subclavian artery. This is one of the primary advantages of approaching these patients through a left thoracotomy – the left subclavian artery can be reimplanted, not just ligated.
Wong and Lee [4] reported a 23-year-old woman who presented with dysphagia and weight loss. She had a right aortic arch, retroesophageal left subclavian artery, and a large Kommerell's diverticulum. The initial procedure was to ligate and divide the left subclavian artery through a left thoracotomy. Her symptoms recurred and at reoperation through a right thoracotomy the Kommerell's diverticulum was resected, with relief of symptoms. This points out the utility of a right thoracotomy for some of these patients as a secondary procedure. The right thoracotomy approach can also be used as a ‘back-up’ if the area of the Kommerell's diverticulum enlarges after left thoracotomy with Kommerell's diverticulum resection and left subclavian artery transfer.
Chun et al. [6] reported 39 cases of congenital vascular rings. In this series, treatment of an important aortic diverticulum (Kommerell's diverticulum) was documented in 19 patients. The aortic diverticulum was excised in nine patients, managed by aortopexy in seven patients, or left in situ in three patients. One boy in that series had persistent severe symptoms due to an untreated aortic diverticulum and underwent subsequent excision of the aortic diverticulum with complete relief of symptoms.
Langlois et al. [5] have reported a large series of patients (468 cases) with aortic arch anomalies. Of 382 patients with isolated thoracic vascular rings, 60 patients had a right aortic arch. Of these 60 patients, two patients were operated on through a right thoracotomy and all other patients were operated on through a left lateral thoracotomy. The ligamentum arteriosum was divided in all patients. The authors recommend that if a Kommerell's diverticulum exists, it should be excised between two clamps. They also recommend dividing the left subclavian artery at the level of the aorta and then reimplanting it into the left common carotid artery in patients with a Kommerell's diverticulum (the number of patients treated in this way is not stated).
Kommerell's diverticulum is an important anatomic vascular structure that can cause recurrent symptoms after vascular ring division. In particular, patients with a right aortic arch, retroesophageal left subclavian artery, and ligamentum arteriosum are at risk of compression of the trachea and/or esophagus from a residual Kommerell's diverticulum if only ligamentum division is performed. In selected patients who are found to have a significant Kommerell's diverticulum after ligamentum division, reoperation with resection of the Kommerell's diverticulum and transfer of a retroesophageal left subclavian artery to the left carotid artery results in relief of symptoms. We are now recommending this as a primary elective procedure for patients with a vascular ring consisting of a right aortic arch, retroesophageal left subclavian artery, left ligamentum arteriosum, and Kommerell's diverticulum serving as a source of the left subclavian artery.
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Footnotes
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Presented at the joint 15th Annual Meeting of the European Association for Cardio-thoracic Surgery and the 9th Annual Meeting of the European Society of Thoracic Surgeons, Lisbon, Portugal, September 16–19, 2001.
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References
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Abstract
1. Introduction
