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Ann Thorac Surg 2002;73:632-633
© 2002 The Society of Thoracic Surgeons

Case report

Bronchial artery aneurysm disguised as a leiomyoma of the esophagus

Stéphane Tringali, MDa, Olivier Tiffet, MD*a, Jean L. Berger, MDa, Jacques Cuilleret, MDa

a Department of General and Thoracic Surgery, CHU Hospital Nord, Saint-Etienne, France

Accepted for publication June 22, 2001.

* Address reprint requests to Dr Tiffet, Service de Chirurgie Générale et Thoracique, CHU Hôpital Nord, ave Albert Raimond, 42055 Saint Etienne Cedex 02, France
e-mail: olivier.tiffet{at}univ-st-etienne.fr


   Abstract
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 Abstract
Introduction
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 References
 
Bronchial artery aneurysm (BAA) is a rare entity with potentially fatal hemorrhagic complications. It is usually diagnosed using a computed tomography scan or angiography in emergency situations (clinical picture of aortic dissection or massive hemoptysis). We report a case of bronchial artery aneurysm where dysphagia was the predominant symptom. Thoracic computed tomography and endoscopic ultrasound seemed to indicate leiomyoma of the esophagus. The final diagnosis revealed a bronchial artery aneurysm.


   Introduction
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 Abstract
 Introduction
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Bronchial artery aneursm (BAA) is a rare entity. It can be classified as intrapulmonary, mediastinal, or both [1]. The principal symptom in cases of mediastinal localization is acute thoracic pain mimicking an aortic dissection or a massive hemoptysis [2–4]. We report a case revealed by a dysphagia and where additional investigation appeared to indicate a benign esophageal pathology.

A 48-year-old woman, without medical history or thoracic trauma, was referred with a history of progressive dysphagia over a 3-month period. Her general status and respiratory functions were normal. Chest roentgenograms were normal. Barium esophagogram showed an extrinsic extramucosal filling defect on the right side of the esophagus at the level of the carina, which suggested a mediastinal pathology. Computed tomography showed a 40-mm diameter round tissue (47 Hounsfields) with regular borders but without calcification, depressing the right side of the esophagus, under the azygos vein. It was not enhanced by the contrast material and showed no adenopathy (Fig 1). Endoscopic ultrasound showed a round, dense hypoechoic lesion on the right side of the esophagus with a fine septum on its superior part and which had common borders with the esophagus. An esophageal leiomyoma was suspected, and we performed an exploratory right thoracotomy. Dissection of the lesion showed the azygos vein surrounding a 4.5 x 3.5 x 1.5 cm structure, joining the right main bronchus and dissociating the muscular fibers of the esophagus. This lesion contained blackish clots. At its upper pole, a right bronchial artery appeared to join up to the lesion. Histopathologic analysis showed a thrombosed saccular aneurysm, without any sign of leiomyoma or neoplasia. The patient’s postoperative recovery was uneventful. Six months later, the patient was asymptomatic and the physical examination was normal.



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  		Fig 1. Chest computed tomographic scan: round tissue picture (47 Hounsfields) depressing the right side of the esophagus.

 

   Comment
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 Abstract
 Introduction
 Comment
References
 
Diagnosis of a BAA with mediastinal localization is usually made in an emergency in the case of massive hemoptysis or acute thoracic pain as shown in 14 cases reviewed by Osada and colleagues [2], and 15 cases reviewed by Kalangos and associates [3]. Hematemesis was described by Shaer and Bashist in 1989 where it was linked to the erosion of a BAA into the esophagus [5]. So the usual clinical presentation of the BAA with mediastinal localization is acute, and the rupture of the aneurysm may cause a fatal hemorrhage. Two cases of asymptomatic aneurysm have been described after a computed thoracic tomography following a pathologic picture on a chest roentgenogram [1]. In our patient, BAA was revealed by a chronic dysphagia (3 months) with no anomalies on the chest x-ray film. The barium esophagogram evoked an extraluminal etiology with an extrinsic compression aspect, which was not enough to affirm a preoperative diagnosis. Computed tomography suggested a benign esophageal pathology, due to its size, its homogeneous aspect, and its regular borders. The hypothesis of vascular origin was not made because the picture did not show any calcification and did not take up the contrast material. From the topography, the most likely diagnosis was leiomyoma; a unique adenopathy of this size was unlikely. The computed tomography diagnosis was difficult because the aneurysm was thrombosed. Nevertheless, endoscopic ultrasound, a key examination for this pathology, showed an unexpected linking angle for a leiomyoma. This examination, exceptionally, can give false results as Konstantakos and colleagues explained in their article, where endoscopic ultrasound suggested a leiomyoma and where the histopathologic analysis revealed an arterial-venous malformation of the esophagus [6]. The surgical exploration rectified the diagnosis, when erosion of the lesion during the surgical dissection released some clots. The diagnosis of a blood cyst or a necrotic adenopathy was canceled, in view of the presence of a bronchial artery at the upper pole of the lesion.

The etiological origin of the BAA is unknown, but bronchiectasis, trauma, a mycotic origin, and Rendu- Osler-Weber syndrome are often associated [2, 3]. We were unable to find any etiology in our observation.

The progressive process of the aneurysm is unknown. Diameter of the aneurysm does not seem to be a risk factor for a rupture [3]. In our observation, the aneurysm’s diameter was bigger than any of the 15 cases reviewed by Kalangos and coworkers and did not show any sign of rupture.

The diagnosis is usually made in an emergency and two different treatments can be proposed: embolization or surgery. Our clinical case with its misleading presentation was treated successfully with surgery. Our observations have shown that a BAA with mediastinal location can be revealed by esophageal compression and can change the usual preoperative evaluation as it appears to be a benign esophageal pathology.


   References
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 Abstract
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 References
 

  1. Fujita J., Akashi K., Kunikase H., et al. A case of bronchial artery aneurysm demonstrating a mass shadow on chest X-ray film. Nippon Kyobu Shikkan Gakkai Zasshi 1991;29:1591-1595.
  2. Osada H., Kawada T., Ashida H., Sodemoto Y., Noguchi T. Bronchial artery aneurysm. Ann Thorac Surg 1986;41:440-442.[Abstract]
  3. Kalangos A., Khatchatourian G., Panos A., Faidutti B. Ruptured mediastinal bronchial artery aneurysm: a dilemma of diagnosis and therapeutic approach. J Thorac Cardiovasc Surg 1997;114:853-856.[Free Full Text]
  4. Yanagihara K., Ueno Y., Kobayashi T., Isobe J., Itoh M. Bronchial artery aneurysm. Ann Thorac Surg 1999;67:854-855.[Abstract/Free Full Text]
  5. Shaer A.H., Bashist B. Computed tomography of bronchial artery aneurysm with erosion into the oesophagus. J Comput Assist Tomogr 1989;13:1069-1271.[Medline]
  6. Konstantakos A.K., Douglas I.W., Abdul-Karim F.W., Lee J.H., Geha A.S. Arteriovenous malformation of the oesophagus disguised as a leiomyoma. Ann Thorac Surg 1995;60:1798-1800.[Abstract/Free Full Text]



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