Ann Thorac Surg 2003;76:1873-1877
© 2003 The Society of Thoracic Surgeons
Original article: general thoracic
Postoperative systemic artery to pulmonary vessel fistula: analysis of three cases
Gregory Riehl, MDa,
Philippe Chaffanjon, MDa,
Gil Frey, MDa,
Carmine Sessa, MDa,
Pierre-Yves Brichon, MD*a
a Service de Chirurgie Thoracique, Hôpital A Michallon, Grenoble, France
Accepted for publication June 4, 2003.
* Address reprint requests to Dr Brichon, Service de Chirurgie Thoracique, Hôpital A Michallon, BP 217, 38043 Grenoble Cedex 9, France
e-mail: pybrichon{at}chu-grenoble.fr
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Abstract
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BACKGROUND: Systemic artery to pulmonary vessel fistulas (SAPVF) occur through pleural adhesions from miscellaneous origin. We report 3 cases of acquired SAPVF that developed late after thoracotomy.
METHODS: There was one pleurectomy for pneumothorax, one sleeve main bronchial resection, and one lower-middle bilobectomy. These SAPVF were discovered 4, 18, and 21 years after surgery.
RESULTS: One patient underwent two unsuccessful embolizations. One patient underwent an unsuccessful attempt at surgical treatment after a previous embolization. Both have persistent SAPVF with minimal clinical discomfort 5 and 13 years later. One patient remains without treatment.
CONCLUSIONS: In the literature 13 cases of SAPVF have been reported after lung resection, pleural drainage, axillary abcess drainage, closed chest trauma, parietal pleurectomy, and talc poudrage. Potential treatments of SAPVF include embolization, resection of pleural adhesion, and artery ligation. The effectiveness of these techniques is uncertain and the follow-up is too short to draw any clear conclusions. Embolization seems to be a useful tool in case of a single afferent artery. Surgical treatment seems to achieve more durable results than embolization but carries a higher risk of bleeding in the case of large SAPVF. Because SAPVF are well tolerated and complications are uncommon, clinical follow-up may be warranted in most cases.
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Introduction
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Most systemic artery to pulmonary vessel fistulas (SAPVF) are congenital. Some SAPVF may also develop as a consequence of tumor or inflammatory processes of pleura or lung or after blunt, open, or iatrogenic chest injuries. We herein report 3 cases of SAPVF that occurred after thoracotomy.
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Material and methods
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Case 1
A 38-year-old man had a fourth-space right axillary thoracotomy for spontaneous pneumothorax with subtotal parietal pleurectomy and stapling of the apex (TA55; USCC, Norwalk, CT) in 1985. Inflammatory thickness of the upper parietal pleura was observed with dense adhesion between the chest wall and the lung. The postoperative course was uneventful. He was referred to us 4 years later because of a new continuous heart murmur. Examination revealed good general appearance of the patient with a continuous "machinery-like" murmur heard in the right axillary and subclavicular areas. Blood pressure was equal on both sides. The chest radiograph showed increased vascular markings in the right lung, predominantly in the lower field, and normal cardiac size. Laboratory tests were normal. Aortic root and right subclavian angiography showed a SAPVF originating from the right internal thoracic artery and the external thoracic branches of the subclavian artery, draining into the right upper pulmonary veins (Fig 1).
In another institution the patient underwent two attempts of systemic embolization using an absorbable gelatin sponge coat without achieving fistula obliteration. The patient had persistent murmur 13 years later and remained asymptomatic at rest and on exertion.
Case 2
In 1979 a 54-year-old woman with a history of primary tuberculosis underwent a left main bronchial sleeve resection through a fifth-space axillary thoracotomy for a severe bronchial stenosis. At the age of 35 she had tuberculosis of the left main bronchus that was medically treated for 1 year. The postoperative course was uneventful except for a pleural effusion at the 30th postoperative day (500 mL, thoracentesis). In October 1997 an expansive left axillary mass was discovered with a continuous murmur in the left subclavicular and axillary areas. A contrast-enhanced spiral computed tomography (CT) scan demonstrated a SAPVF under the thoracotomy scar between the left internal and external thoracic arteries, intercostal and diaphragmatic arteries, and the left pulmonary veins (Fig 2).
A decision was made to perform surgery on this patient and embolization of the branches of the internal and external thoracic arteries was performed on the day before surgery. Primary trunk ligation of the fourth, fifth, sixth, and seventh intercostal arteries was performed through a posterolateral thoracotomy. But any attempt to enter the thorax by several intercostal spaces was aborted because of severe bleeding from arterial collateral network of the periosteum. The surgeon decided not to carry on with the operation. After 5 years of follow-up this SAPVF has no clinical consequence and seems to have a slightly decreased shunt flow, perhaps because of the embolization.
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Fig 2. Contrast-enhanced spiral computed tomography scan showing the parietal inflow artery and the upper left pulmonary outflow vein. Note the hypertrophy of both lower vein and pulmonary artery in the fissure, the dilatated internal thoracic artery, and a tortuous parietal artery in front of the scapula.
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Case 3
A 37-year-old man underwent a lower-middle bilobectomy for bronchiectasis in 1979. He was referred in April 2001 for cough, asthenia, hemoptysis, and right-side chest pain. The chest radiograph showed no abnormality on the right side. The contrast-enhanced spiral CT scan revealed a SAPVF between one single intercostal artery and the pulmonary veins in the area of the posterolateral thoracotomy scar (Fig 3).
The cardiac index was 3.3 L · min-1 · m-2 (normal value; 2 L · min-1 · m-2). The patient refuses any treatment at the present time.
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Fig 3. Contrast-enhanced spiral computed tomography scan. The systemic artery to pulmonary vessel fistula is inside a thickened pleura (black arrow).
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Comment
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Systemic artery to pulmonary vessel fistulas are uncommon abnormal communications between systemic arteries (except bronchial arteries) and pulmonary arteries or veins. The first case was described in 1947 by Burchell and Clagett [1]. In 1990 Remy-Jardin and Remy [2] called this SAPVF the "3rd arterial circulation of the lung or non bronchial systemic arterial circulation." All SAPVF develop in neoplastic or inflammatory diseases causing pleural adhesions that allow penetration of nonbronchial systemic arteries into lung parenchyma. The most frequent afferent arteries involved in the SAPVF are the internal thoracic and intercostals. The fistulas may also arise from abnormal aortic branches and from subclavian, axillary, diaphragmatic, mediastinal, and coronary arteries. The SAPVF outflow is to the pulmonary artery or the pulmonary vein or both. Fifty percent of SAPVF are congenital and occur in the presence of cardiopathy or pulmonary artery hypoplasia [3, 4]. Noncongenital SAPVF develop in patients with cancer or with various infectious or inflammatory disease of the pleura and the lung including tuberculosis, mycosis, pneumonia, lung abscess, Takayashu's disease, or after trauma [5]. In these cases abnormal circulation develops as a consequence of a downstream blockage of the involved vessels (especially the internal thoracic artery) or when the pleural inflammatory process causes chest wall or mediastinal collateral circulation. Since the first case described by Cox and coworkers [6] in 1967 we found 13 other cases of traumatic or iatrogenic SAPVF in the literature (Table 1).
Four cases occurred after lung resection [7–10], 3 cases were located at the site of isolated chest tube drainage [6, 11, 12], 2 cases developed after blunt chest trauma [13, 14], 2 cases after percutaneous drainage of axillary abscess [15], 1 case after talc pleurodesis [16], and 1 case after parietal pleurectomy for pneumothorax [17]. Delay in diagnosis ranges from 1 to 20 years.
The SAPVF is usually asymptomatic and discovered during a routine physical examination. Symptomatic patients present with dyspnea, congestive heart failure, or hemoptysis [18]. Chest radiograph may reveal an increased lung vascularization, parenchymal infiltration, or rib notching if intercostal arteries are involved. Contrast-enhanced spiral CT scan is a useful tool for diagnosis but selective angiography allows better anatomic mapping of afferent arteries if embolization is planned. The natural outcome of SAPVF is not yet well known. Complications described include hemoptysis [4, 17] or congestive heart failure [14]. Other potential complications such as pulmonary hypertension, endocarditis, or spontaneous rupture have never been described. A SAPVF may be managed by embolization, resection, or observation.
Embolization
Three cases of SAPVF of a single afferent artery have been reported with two successful results on short follow-up (2 months). The third case was unsuccessfully treated: a first attempt of embolization of the internal thoracic artery led to immediate success but the thoracic murmur returned after 10 days. Surgical ligation of three branches of the subclavian artery was performed without success. Thereafter a second embolization in the same area was successful. However the murmur again returned 2 years later. In this latter case, as in our first and second cases, the SAPVF were complex with many afferent arteries. Thus embolization seems to be an efficient treatment if the SAPVF has only one afferent artery. Nevertheless published results in the literature will have to be supported by a longer follow-up.
Surgery
Five attempts have been published, one of them associated with embolization as previously described. The treatment consisted of isolated ligation of inflow arteries in 2 cases, surgical division of pleural adhesions with or without excision of the involved lung parenchyma in 2 cases, and a combination of the two procedures in 1 case with the use of a Mersilene mesh (Ethicon-Johnson & Johnson, New Brunswick, NJ) to definitely separate the lung from the chest wall. There were two successful results: one isolated ligation and one combined procedure using Mersilene mesh with a 15-month follow-up. There were three failures: two isolated ligations with previous embolization in one case and one surgical division of the pleural adhesion. We found one death as a result of severe bleeding during the surgical treatment of a congenital SAPVF [19]. Severe bleeding during surgery forces an end to surgery as reported in the literature [1, 20] and as in our described case 2. Surgery may be attempted if the SAPVF has few inflow arteries. Surgical results seem to be more durable than embolization. However if the SAPVF has multiple arterial input, there is a risk of severe bleeding. The use of a prosthesis to definitively separate the lung from the chest wall may be considered [6]. The use of a polyfluorethylene (Gore-tex) prosthesis seems theoretically better than Mersilene mesh in avoiding postoperative pleuro-parenchymatous inflammatory adhesions.
Observation
Five cases have had no attempt at treatment, with no death or worsening of clinical symptoms. The first 2 patients of our series have persistent SAPVF with minimal clinical discomfort at 5 and 13 years follow-up.
The management of SAPVF is difficult and controversial. For many authors the occurrence of hemoptysis seems to warrant surgical treatment. However surgery is difficult, with a high bleeding risk directly related to the number of inflow arteries. Owing to the long delay in the development of the SAPVF, the cases published in the literature have too short a follow-up to draw any clear conclusion on the most appropriate and effective management. As most SAPVF are well tolerated and complications are uncommon, simple observation may be warranted in some cases, with careful follow-up.
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Acknowledgments
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The authors thank Dr Frederic W. Grannis, Jr for help with the language.
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References
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Abstract
Introduction
