Ann Thorac Surg 2005;79:e29-e31
© 2005 The Society of Thoracic Surgeons
Case reports
Nontraumatic Chest Wall Systemic-to-Pulmonary Artery Fistula
Hideki Itano, MDa,
Sang Lee, MDa,
David M. Kulick, MDa,
Mark D. Iannettoni, MDa,
David M. Williams, MDb,
Mark B. Orringer, MDa,*
a Department of Surgery Ann Arbor, Michigan
b Department of Radiology, University of Michigan, Ann Arbor, Michigan
Accepted for publication January 10, 2005.
* Address reprint requests to Dr Orringer, 1500 E Medical Center Dr, Taubman Center, Box 0344, Ann Arbor, MI 48109-0344 (E-mail: morrin{at}umich.edu).
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Abstract
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A congenital chest wall systemic-to-left pulmonary artery fistula fed by the left internal mammary and left gastric arteries in a 31-year-old man is reported. Attempted sclerosis was complicated by rupture of the communicating vessel, hemothorax, and deep vein thrombosis of the legs. Fistula resection and pulmonary decortication were successfully performed.
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Introduction
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Congenital systemic-to-pulmonary artery (S-PA) communications other than those associated with patent ductus arteriosus, pulmonary sequestration, and congenital heart disease are rare. We believe that only 19 cases of congenital S-PA fistulas have been reported (Table 1), and therefore this represents case report 20.
A 31-year-old asymptomatic man with no history of trauma, pneumonia, or pleural disease, and a normal physical examination had an abnormal routine chest roentgenogram. A chest computed tomographic scan showed a 4.5 cm chest wall mass adjacent to the lingula and no associated pleural or pulmonary disease. Pulmonary arteriography showed no pulmonary arteriovenous malformation. Systemic artery injections showed hypertrophied left internal mammary and gastric arteries supplying a vascular malformation that emptied into an anomalous vessel communicating with the left descending pulmonary artery (Fig 1A–C). Pulmonary artery pressure was 25/13 mm Hg (mean, 18 mm Hg). Sclerosis of the vascular malformation was undertaken. Catheters were placed distally in the left internal mammary and gastric arteries in the event that rapid coil occlusion of the feeding vessels might be needed. The communicating vessel was then cannulated from the pulmonary artery side, and an occlusion balloon (Boston Scientific Corp, Natick, MA) was inflated prior to retrograde injection of a sclerosant. However, balloon inflation was complicated by rupture of the vessel. Multiple embolization coils were then deposited through the catheters in the two feeding vessels to reduce arterial inflow. The patient was then stabilized. A chest tube was placed to treat the resulting clotted left hemothorax. Four days later, deep venous thrombosis of the right femoral and left infrapopliteal venous system necessitated inferior vena cava filter placement. The need for anticoagulation, concern about re-bleeding from the injured pulmonary artery, and persistence of the hemothorax prompted evacuation of the hemothorax and resection of the vascular malformation.
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Fig 1. Angiography of chest wall systemic-to-pulmonary artery fistula (S-PAF). (A) Left internal mammary arteriography. (B) Left gastric arteriography, arterial phase. (C) Left gastric arteriography, venous phase. The dominant feeding vessels were the left internal mammary (IMA) and left gastric arteries (LGA). There was a small contribution from four intercostal and the left lateral thoracic arteries. The outflow pathway was through a channel into the left pulmonary artery (PA).
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Through a left seventh interspace posterolateral thoracotomy, clot evacuation and pulmonary decortication were performed. The tortuous left internal mammary artery entering the mass in the antero-medial costophrenic angle adjacent to the adherent lingula was suture-ligated. A large vessel from the malformation into the lingula was divided with a surgical stapler. The vascular mass was then dissected from the mediastinum, the diaphragm, and the anterior chest wall. Intraoperative blood loss totaled 1,850 mL, and 4 units of blood were transfused. The resected mass measured 6.0 x 3.5 x 1.0 cm. Pathology confirmed an arteriovenous malformation. The patient's postoperative course was uneventful, and he remains well 3 months later.
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Comment
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Systemic-to-pulmonary artery fistulas may be classified as either congenital or acquired. Nineteen congenital S-PA fistulas have been reported (Table 1). Congenital S-PA fistulas may result from functional activation by genetic or external influences of relatively dormant pre-capillary and postcapillary bronchopulmonary arterial anastomoses found in the normal lung [1, 2].
Unlike right-to-left shunts with associated cyanosis, digital clubbing, or polycythemia, these left-to-right shunts are often asymptomatic and discovered by chance because of a chest wall bruit or rib notching or an abnormal shadow on screening chest roentgenograms [3]. Selective angiography delineates the anatomy and location of the lesion, feeding, and draining vessels, and the extent of pulmonary parenchymal involvement. The S-PA fistulas may occur within or outside the lung parenchyma, in the chest wall, or in the diaphragm [4]. Pulmonary angiography may show early washout of contrast from involved pulmonary artery segments [3]. The vessels most frequently involved are internal mammary and intercostal arteries. Of the 19 reported congenital S-PA fistulas, 14 (73.7%) involved the internal mammary arteries, and 3 (15.8%) involved the intercostals (Table 1). The average age at diagnosis of congenital S-PA fistulas is 22.4 years, and the male-to-female ratio is 3.75 to 1. The right lung has been involved in 10 cases, the left in 7, and both in 1.
Because their natural history is unknown, and patients are usually asymptomatic, precise indications for intervention have not been established [3]. Potential complications are infectious (bacterial seeding), hemorrhagic (enlargement and rupture), and cardiac (pulmonary artery hypertension or congestive heart failure) [5].
Definitive treatment of S-PA fistulas is resection of the fistula and involved lung parenchyma. Recurrence has been reported with ligation of feeding vessels alone. In 10 of the 19 reported congenital S-PA fistulas, an enlarging shunt prompted surgery. Angiographic embolization was used in two congenital [6] and three acquired cases, some of which recurred and needed repeated embolization [7]. In our patient, attempted sclerosis of the fistula resulted in significant morbidity as previously described. Balloon occlusion of the draining vessel, which is a useful adjunct to achieve temporary stasis during retrograde injection of a sclerosant to treat systemic AVMs, ruptured the communicating vessel in this patient. It is possible that the anomalous vessel was not as robust structurally as a systemic vein. Because the vessel was extrapulmonary and outside the inferior pulmonary ligament, poorly contained hemorrhage into the pleural space ensued. In retrospect, it might have been beneficial to reduce arterial inflow by means of coils and glue before inflating the occlusion balloon. This case suggests that S-PA fistulas may be more hazardous to treat percutaneously than systemic AVMs, and that the possible benefits of direct surgical intervention with resection of the fistula should be carefully considered.
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References
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- Robinson LA, Sabison DC. Syndrome of congenital internal mammary-to-pulmonary arteriovenous fistula associated with mitral valve prolapse Arch Surg 1981;116:1265-1273.[Abstract/Free Full Text]
- Brundage BH, Gomez AC, Cheitlin MD, Gmelich JT. Systemic artery to pulmonary vessel fistulas: report of two cases and a review of the literature Chest 1972;62:19-23.[Abstract/Free Full Text]
- Greenberg BH, Stasikowski JJ, Harrison CE, et al. Epicardial artery-pulmonary artery fistula AM J Cardiol 1968;22:893.[Medline]
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- Hirsch M, Maroko I, Gueron M, Goleman L. Systemic pulmonary arteriovenous fistula of traumatic origin: a case report Cardiovasc Intervent Radiol 1983;6:160-163.[Medline]
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Abstract
Introduction
