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Rupture of aorta arch aneurysm into the lung with formation of pseudoaneurysm
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     Department of Cardiovascular and Thoracic Surgery, West-China Hospital, Sichuan University, Guoxuexiang, No.37, Sichuan, Chengdu 610041, China

    Abstract

    Objective: To improve the early diagnosis and avoid preoperative misdiagnosis, we discuss some of the diagnostic problems related to thoracic saccular aneurysm rupture into the lung. Methods: A case report is presented. We report on a patient with rupture of aorta arch aneurysm into the lung with formation of pseudoaneurysm accompanied with left upper lobe atelectasis, who simultaneously underwent a left upper lobectomy, aneurysmectomy and aorta arch neoplasty. Results: The few cases described in the literature are reviewed to discuss the difficulties of differential diagnosis of left lung mass. The diagnosis methods and operation treatment of the aorta aneurysm are discussed. Conclusions: Angiography and CT are effective diagnostic methods of aorta aneurysm. These data suggest that it is an effective method of aneurysmectomy with/without left pneumonectomy or lobectomy to treat aneurysm of thoracic aorta.

    Key Words: Aneurysm of thoracic aorta; Left lung; Operation

    1. Introduction

    The key causes of death in rupture of thoracic aneurysm are cardiac tamponade and hemorrhea [1,2]. Hemoptysis and hoarseness as the presenting symptoms of the aorta aneurysm rupture into the lung often causes diagnostic difficulties. The most important differential diagnosis of the aorta aneurysm rupture into the lung is lung cancer invading the aorta. Here, we report on a patient with an aorta arch aneurysm rupture into the left upper lung accompanied with formation of pseudoaneurysm misdiagnosed left lung cancer invading the aorta, who simultaneously underwent a left upper lobectomy, and aneurysmectomy and surgical repair under extracorporeal circulation.

    2. Case report

    A 65-year-old woman was admitted to our hospital because of hemoptysis for 7 days. Eight months earlier, she had presented to another hospital with productive cough, hemosputum and hoarseness. She was treated with antibiotics and non-steroidal anti-inflammatory drugs, but her bloody sputum and hoarseness did not resolve. Presently, contrasted computed tomography of the chest showed a well-defined different density left upper lobe mass, which invaded the side wall of the aorta arch and the aorta arch rupturing into the mass with formation of hematoma (Fig. 1 a,b,c). The mass in the left upper lobe was significantly larger than that at eight months and also at four months earlier (Fig. 2 a,b). Physical examination was normal except for the presence of respiratory sound in the left lung which was low. Cervical lymph node size was not significant on admission. Results of sputum cultures were negative. The electrocardiogram showed sinus rhythm. A fiberbronchoscope revealed that the tracheal cavity and bronchial lumen were normal, but left vocal cord paralysis was found. We considered a diagnosis of lung cancer of the left upper lobe invading the wall of the aorta. She had no history of smoking or any significant pulmonary disease. No history of trauma or anticoagulation was present.

    The patient first underwent a left upper lobectomy, and aneurysmectomy and surgical repair under extracorporeal circulation that revealed a mass, measuring 10x9x8 cm, abutting the wall of aorta arch and descending aorta, a 1.5 cm rupture was found in the anterior wall of aorta arch. The aorta aneurysm ruptured into the left upper lung with formation of pseudoaneurysm. Histologically, the specimens of the left upper capsular space and the aorta wall were composed of hemorrhage, necrosis tissue, and fibroplasias. These findings indicated that the rupture of aorta arch aneurysm into the lung with the formation of pseudoaneurysm in the left superior lobe of the lung. The patient had an uneventful postoperative course and also on follow-up studies, 10 months later. A reconstructed CT scan documented complete exclusion of the aneurysm and a normal size aorta.

    3. Discussion

    Hemoptysis has numerous causes; most cases are associated with a chronic infectious process, such as tuberculosis and bronchiectasis, respectively [3]. Hemoptysis occurs in more than 50% of lung cancer cases, and can be the result of direct invasion of the bronchial arteries, tumor manipulation during diagnostic fiberoptic bronchoscopy, or distal ischemia and avascular necrosis [4]. Direct aortic invasion by an infectious process has also been described as a cause of hemoptysis [5].

    Few case reports of aneurysm rupturing into the lung presented with hemoptysis. Other symptoms typically have been reported including chest or back pain, cough, dyspnea, and hemoptysis [6,7]. An aortic aneurysm or dissection that ruptures into the lung parenchyma or erodes into a bronchus can lead to acute and massive hemoptysis. Aorto-bronchopulmonary fistulas account for 85% of cases of hemoptysis that occur in conjunction with a descending thoracic aneurysm. The bleeding is frequently massive, but can also be relatively light because of severe adhesion into the attached lung tissue [8]. However, presenting with hemosputum and hoarseness is rare. Investigations revealed a thoracic aortic aneurysm compressing the left recurrent nerve; thus, the diagnosis of Ortner's syndrome, i.e. cardiovocal syndrome, could be established and at the 6-month follow-up visit the hoarseness had partly resolved.

    Aortic arch aneurysms are most often asymptomatic, but can present with symptoms caused by compression or acute aneurysm expansion such as upper chest or shoulder pain, and hoarseness. In the present case, the patient had an increasing enlargement mass of left upper lobe suggestive of lung cancer, especially because of oral dry cough, hemoptysis and hoarseness, which are the most common symptoms of the lung cancer. The hemoptysis was thought to be the result of invasion of the bronchial tree by the cancer and the preoperative CT showed the left upper lobe mass eroding into the aorta arch anterior wall and fistula formation with lung parenchyma. Therefore, it is diffcult to diagnose between a lung cancer invading the aorta and a rupture of arota arch aneurysm extending into the lung. Aortic arch aneurysm misdiagnosed as lung carcinoma. We were surprised to find that the hemoptysis was actually caused by an aortic aneurysm rupturing into the lung.

    Computed tomography (CT), magnetic resonance imaging (MRI) and angiography are usually the most valuable methods of diagnosing an aneurysm; these techniques have proven to be unsatisfactory in distinguishing between aneurysm and lung cancer [9,10]. Angiography is a more sensitive and accurate means of diagnosing aneurysm, especially rupture of aorta aneurysm [11]. However, they were not sufficient to reveal the exact nature of the lesion in our patient, because the aortic arch aneurysm rupturing into lung formatted a small cystic pseudoaneurysm and limited the center of the lung mass. In this case report it is unique in that the aneurysm had ruptured through fibrous adhesions into the visceral pleura and lung; the blood lodged in the lung parenchyma instead of the pleural space. The fibrous tissue attachment between the lung and aorta was the result of an old adhesive pleuritis for which pathological evidence was supplied by the specimen studied. Unfortunately, angiography performed before preoperation could not identify the aortic arch aneurysm in the left upper lobe. Correct diagnosis was made by exploratory thoracotomy.

    Aortic aneurysms are at an increased risk of rupture, embolization, or thrombosis. Hence, they should be considered for surgical repair. The surgical mortality rate in patients with ruptured thoracic aneurysms is high compared with that of patients undergoing elective resection [12]. However, in a large retrospective analysis, recommended treatment is surgical treatment, early and long-term results of surgery for thoracic aortic aneurysm (of all types) demonstrated best outcomes in patients with operation [13–16]. These data suggest that it is an effective method of aneurysmectomy with/without left pneumonectomy or lobectomy to treat aneurysm of thoracic aorta.

    This case of a 65-year-old woman with an aorta arch aneurysm rupturing into the lung is unusual in that the patient presented with hemoptysis and hoarseness.

    Appendix A. ICVTS on-line discussion

    Author: Carlos A. Mestres (Cardiovascular Surgery, Hospital Clinico, University of Barcelona, Barcelona, Spain)

    eComment: It is truly a very uncommon situation to find a case of lung cancer with invasion and erosion of the aorta, however, it can happen as shown by the authors. I found the solution quite elegant and it reinforces the fact that sometimes, extended surgery can be performed looking for oncological criteria of radicality. Follow-up up to 10 months seems to be satisfactory but the reader would like to see up to when the patient was followed to justify this more radical approach.

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