Ann Thorac Surg 2002;73:312-313
© 2002 The Society of Thoracic Surgeons
Case report
Dyspnea lusoria: compression of the pulmonary artery by a Kommerell’s diverticulum
Mahmoud B. Malas, MDa,
Mark L. Barr, MDa,
Vaughn A. Starnes, MDa,
Shelley Shapiro, MD, PhDa,
Suzanne Palmer, MDa,
Daniel S. Schwartz, MD*a
a Department of Cardiothoracic Surgery, University of Southern California, Keck School of Medicine, Los Angeles, California, USA
Accepted for publication April 26, 2001.
* Address reprint requests to Dr Schwartz, Department of Cardiothoracic Surgery, University of Southern California School of Medicine, 1510 San Pablo St, Suite 415, Los Angeles, CA 90033, USA
e-mail: dschwartz{at}surgery.usc.edu
 |
Abstract
|
|---|
We report a rare case of Kommerell’s diverticulum presenting as dyspnea on exertion. After careful physical examination, selective imaging tests demonstrated a significant flow abnormality in the left pulmonary artery caused by an aneurysm of the descending aorta in the area of the ligamentum arteriosum.
|
Introduction
|
|---|
In 1936, Kommerell described a diverticulum of the descending aorta at the ligamentum arteriosum caused by the persistence of the posterior segment of the fourth aortic arch. This anomaly now bears his name [1]. Kommerell’s diverticulum is rare, occurring in only one-half of 1% of the population [2]. Symptomatic Kommerell’s diverticulum is even less common and is usually caused by an associated aneurysm. Compression of the esophagus with dysphagia is the most common symptom [3]. Dyspnea by compression of the airway is less common. We report dyspnea caused by compression of the pulmonary artery from an aneurysm of Kommerell’s diverticulm.
The patient was an otherwise healthy 39-year-old woman who presented with a 2-month history of dyspnea on exertion. Chest roentgenogram demonstrated a 4.4-cm mass in the left hilar region. Computerized tomography scan showed a 3.6-cm thoracic aneurysm that appeared to be coming between the aorta and left pulmonary artery causing significant narrowing of the later. The parasternal short axis view of the echocardiogram demonstrated flow turbulence across the left pulmonary artery. Continuous Doppler echocardiography demonstrated increased velocity of 1.9 meters per second (normal 0.7 to 0.9 m/s) indicating a significant narrowing of the artery (Fig 1).
Magnetic resonance angiogram of the aorta showed a 3.5 x 3.0 x 3.0-cm aneurysm arising from the descending aorta in the area of the ligamentum arteriosum with almost complete compression of the left main pulmonary artery (Fig 2).
The trachea and both mainstem bronchi were patent without external compression (not shown on this cut).
View larger version (33K):
[in this window]
[in a new window]
|
Fig 1. Continuous Doppler across the left pulmonary artery showing an increased velocity of 1.9 meters per second (m/s). Normal range is 0.7 to 0.9 m/s.
|
|
View larger version (80K):
[in this window]
[in a new window]
|
Fig 2. Magnetic resonance angiogram demonstrating the aneurysm (A) originating from the aorta (Ao), compressing but not communicating with the left pulmonary artery (arrow). (H = head; MPA = main pulmonary artery.)
|
|
A lateral muscle sparing thoracotomy was performed through the fourth intercostal space. Single lung ventilation with left atrial to left femoral artery bypass was utilized. The aneurysm appeared to compress and nearly occlude the left pulmonary artery. There was no obvious compression of the airway. A 20-mm hemashield interposition graft was used to replace the aneurismal aorta. The postoperative course was uncomplicated. The patient continues to do well 1 year following the operation, with complete resolution of her symptoms.
|
Comment
|
|---|
Kommerell’s diverticulum is a rare congenital abnormality of the descending aortic arch occurring in only 0.5% of the population [2]. The diverticulum is usually located at the origin of an aberrant subclavian artery. It is usually found incidentally on chest roentgenogram and is often misdiagnosed as a mediastinal tumor. On the rare occasion when the diverticulum produces symptoms, it is usually due to an associated aneurysm [4]. The most common symptom is dysphagia caused by compression of the esophagus. Shannon was the first to describe a case of esophageal obstruction by an isolated Kommerell’s diverticulum [5]. In 1794, Bayford termed this phenomenon dysphagia lusus natura or dysphasia by a freak of nature; it has since come to be known as dysphagia lusoria [6, 7]. Dyspnea by compression of the airway is less common.
Aneurysms of Kommerell’s diverticulum are at significant risk of rupture regardless of whether they are symptomatic or not. Observation with 6-month interval computed tomographic scan is probably appropriate in poor-risk patients with aneurysms less then 4 cm in greatest dimension [8, 9]. Otherwise, these aneurysms should be repaired when discovered because of the high risk of rupture and death [8, 9].
In summary, aneurysm of Kommerell’s diverticulum is a rare anomaly that can produce significant compression of the surrounding mediastinal structures. We report an unusual case of dyspnea lusoria caused by compression of the left pulmonary artery by an aneurysm originating at Kommerell’s diverticulum.
|
References
|
|---|
-
Kommerell B. Verlagernung des osophagus durch eine abnorm verlaufende arteria subclavia Dextra (arteria lusoria). Fortschr Geb Rontgenstr Nuklearmed Erganzungsband 1936;54:5905.
-
Walker T.G., Geller S.C. Aberrant right subclavian artery with a large diverticulum of Kommerell: a potential for misdiagnosis. AJR Am J Roentgenol 1987;149:477-478.[Free Full Text]
-
Brown D.L., Chapman W.C. Dysphagia lusoria: aberrant right subclavian artery with a Kommerell’s diverticulum. Am Surg 1993;59:582-586.[Medline]
-
Felson B. Ruptured anomalous right subclavian artery: aneurysm or diverticulum?. Semin Roentgenol 1989;24:121-126.[Medline]
-
Shannon J.M. Aberrant right subclavian artery with Kommerell’s diverticulum. J Thorac Cardiovasc Surg 1961;41:408-411.
-
Bayford D. An account of a singular case of obstructed deglutition. Memoirs Med Soc Lond 1794;2:275–86.
-
Stoney W.S., Alford W.C., Burrus G.R., Thomas C.S., Jr Aberrant right subclavian artery aneurysm. Ann Thorac Surg 1975;19:460-467.[Abstract]
-
Verkroost M.W., Hamerlijnck R.P., Vermeulen F.E. Surgical management of aneurysms at the origin of an aberrant right subclavian artery. J Thorac Cardiovasc Surg 1994;107:1469-1471.[Abstract/Free Full Text]
-
Cooley DA. Surgical treatment of aortic aneurysms. Philadelphia: WB Saunders, 1986:175–84.
Top
Abstract
Introduction
