Ann Thorac Surg 2005;80:365-370
© 2005 The Society of Thoracic Surgeons
Review
Right Pulmonary Artery to Left Atrium Communication
Ujjwal K. Chowdhury, MCha,*,
Diplomate NB,
Shyam S. Kothari, DM, FACCb,
Balram Airan, MCha,
K. Ganapathy Subramaniam, MSa,
Panangipalli Venugopal, MCha
a Department of Cardiothoracic and Vascular Surgery, Cardiothoracic Centre, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, India
b Department of Cardiology, Cardiothoracic Centre, All India Institute of Medical Sciences, Ansari Nagar, New Delhi, India
* Address reprint requests to Dr Chowdhury, Department of Cardiothoracic and Vascular Surgery, All India Institute of Medical Sciences, Ansari Nagar, New Delhi 110029, India (Email: ujjwalchow{at}rediffmail.com).
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Abstract
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This is a collective review of 59 published cases of right pulmonary artery to left atrium communication in the English-language literature. In this article, we review the literature on the clinical presentation, anatomic variations, diagnostic features, and management considerations of previously reported cases. Two-dimensional echocardiography and cardiac catheterization including selective angiography provided the necessary diagnostic information and defined the anatomy before surgery in all patients. Various management options including medical, surgical, and transcatheter closure have been reported. The lesions have been classified into four types (I, II, III, and IV) that have a bearing on the appropriate surgical approach and techniques of repair that are discussed in the text. Use of extracorporeal circulation may be considered in selected instances. The operative mortality rate during the earlier era (1950 to 1979) was high, at 22%. More recently, the mortality rate has approached zero. An individualized surgical approach depending on the type of surgical connections is recommended.
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Introduction
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Right pulmonary artery to left atrium communication is a rare congenital entity of both clinical and surgical fascination [1–9]. The disease entity should be suspected when the following combination of symptoms and signs coexist: central cyanosis, clubbing of fingers, exertional dyspnea, silent precordium, normally split second heart sound, nonspecific murmur over left or right axillary region, and an abnormal roentgenographic density in the right or left pulmonary hilum [1–10]. The exact diagnosis is relatively easy to make by contrast echocardiography and selective angiocardiography, provided the possibility is entertained, and complete cure can be obtained by surgery. This review takes into account variations in anatomy that have a bearing on the management strategies.See page 336
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Incidence
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The true incidence of right pulmonary artery to left atrium communication is unknown. It is an uncommon congenital or acquired cardiopulmonary vascular anomaly [1–9]. The majority of these fistulas are congenital; however, a posttraumatic variety has been infrequently described in adults [6]. Mention of this abnormality in contemporary cardiology textbooks is uncommon.
The first case of right pulmonary artery to left atrium fistula was operated by Blalock and described by Friedlich and coauthors in 1950 [1]. Literature search to date revealed 59 cases with excellent reviews discussing mainly the clinical features, anatomical aspects, and pathogenesis of this entity (Table 1) [1–9].
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Clinical Presentation
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The age of the patient at initial diagnosis ranged from 1 day to 60 years, and the male-to-female ratio was approximately 3:1. It was not possible to identify any regional or ethnic predominance [1–9, 11–16]. From a review of cases (Table 1), one can readily see that this malformation appears to have two clinical forms of presentation: a very severe form in the newborn period (n = 10, 15.8%), and a milder, less lethal form in older patients. The severity of symptomatology appears to be dependent on the magnitude of the right-to-left shunt [1–9, 11–16]. Central cyanosis, decreased arterial oxygen saturation, tachypnea, and respiratory distress often complicate the infant’s intensive care unit course [5, 7–9, 11–13]. In newborns, large shunts can cause severe congestive cardiac failure necessitating emergency surgical intervention [5, 7–9, 11–13]. Cyanosis with exertional dyspnoea as well as clubbing of the fingers and toes are often the findings in older children [2, 3, 14–24]. By 1 year of age, only 20% had been diagnosed. Eight of 10 newborns needed urgent surgical intervention, 1 newborn underwent successful catheter-based coil closure, and 1 infant responded favorably to medical management [7, 11]. Only 4 newborn patients survived [5–7, 9, 11–13].
Causes of death were probably related to additional cardiac lesions, which mostly included patent ductus arteriosus or patent foramen ovale or both. Probably, patency of the ductus anteriosus was important, causing a large volume load of the left ventricle and leading rapidly to cardiac failure [5, 7–9, 11–13]. Older patients usually have a milder form of the disease and are not symptomatic until adolescence or adulthood [2, 3, 14–16]. Central cyanosis and clubbing are seen in about 60% of patients. Twenty percent of patients have neurologic symptoms [2, 5, 7–9, 14–18].
Untreated, pulmonary arteriovenous fistulas may lead to pulmonary edema with pulmonary hypertension [1–9, 13–16]. Because the pulmonary artery communicates directly with the left atrium, bypassing the pulmonary filter function, the patient may present with cerebral or systemic embolization or with cerebral abscesses [2, 15, 16, 18–24]. Other reported complications are endarteritis, infective endocarditis, and aneurysmatic growth of the fistula, with the risk of fatal rupture [1–9, 15, 18–24].
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Diagnosis
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Diagnosis of this anomaly is a rewarding clinical experience. Laboratory examination shows arterial hypoxia and polycythemia [1–9, 13–16]. The systemic arterial oxygen saturation and the hematocrit in the reported patients ranged from 65% to 82% (average 72%), and 62% to 80% (average 76%), respectively [1–9, 11–13, 16, 17–22].
Electrocardiography may reveal left atrial enlargement and left ventricular hypertrophy with left axis deviation. Only patients with a large right-to-left shunt have the above features [1–9, 11–13, 15–18].
Chest roentgenography may reveal cardiomegaly with left ventricular hypertrophy and poorly filled pulmonary arteries in extreme cases. The abnormal right paracardiac silhouette on chest roentgenogram seems to be a rather subtle but constant finding in patients with right pulmonary artery to left atrium communication [10]. Zanchetta and coworkers have reported the usefulness of computed tomographic imaging of the chest to visualize the angioarchitecture of a large fistula-type connection between the right inferior pulmonary artery and the right posterior left atrial wall [14].
Although the combination of central cyanosis, oligemic lung fields, and electrocardiographic evidence of left ventricular preponderance suggests a diagnosis of tricuspid atresia or hypoplastic right ventricle, an echocardiogram will rule out these diagnoses by showing a normal right ventricle [15]. Only a few echocardiographic studies have been reported showing the indirect effects of the malformation: large pulmonary artery, left atrium, and left ventricle [1–9, 11–13, 16, 18–23]. An aneurysmal-like pouch behind the left atrium has been described [7]. Pulsed Doppler, reported for the first time by Jimenez and coworkers [9] has helped to confirm the diagnosis by showing diffuse turbulence in the left atrium that could be followed to the aneurysmal structure and the anomalous pulmonary artery connection. Contrast echocardiography will definitely help to confirm a pulmonary artery to left atrial or pulmonary venous fistula, the pathognomonic finding being early appearance of dense contrast in the left atrium (within two to three cardiac cycles) when contrast is injected into the arm veins [7].
There is an extensive literature devoted to the hemodynamic and angiographic assessment of these patients [1–9, 11–14, 19–25]. Cardiac catheterization findings include oximetry demonstrating a right-to-left shunt at the atrial level with systemic arterial desaturation. Pulmonary artery pressures are usually normal; however, in the neonate they may be elevated. Angiography is definitive, demonstrating the ectatic proximal right pulmonary artery and the dilated fistula vessel draining into the left atrium.
The disease entity should be considered as part of the differential diagnosis when such conditions as these are found: a left superior vena cava that drains into the left atrium, an unroofed coronary sinus, or an atrial septal defect with elevated right atrial pressure and a right-to-left shunt [2, 3, 15, 16].
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Embryogenesis
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No definite embryogenetic explanation has been described for this anomaly. In fact, the disease spectrum includes a heterogenous group of cases, and there may be a different embryonic development in each of this anatomic variation [2, 18–20, 26].
Some investigators have hypothesized that this type of lesion may result from a fistula which, during early embryonic development forms between the pulmonary artery and one of the main pulmonary veins. Later, the large pulmonary vein becomes incorporated into the wall of the left atrium during atrial enlargement [19, 26]. These authors believed that the fistula represented the original vascular connections of the accessory lobe of the right lung. Pulmonary vein anatomy was not described in their report [19, 20].
This embryogenetic explanation is not shared by Tuncali and Aytac [27] because all pulmonary veins of their patient drained into the vascular dilatation of the fistula before they entered the left atrium. Some authors consider this anomaly as a variant of pulmonary ateriovenous fistula; however, unlike pulmonary arteriovenous fistulas, pulmonary-to-left atrium fistulas do not involve direct communication between an artery and a vein [1, 18]. These authors have postulated that some of the right pulmonary artery–left atrial communications are the result of agenesis of the lobe of the lung and consequent absence of the pulmonary capillary bed [18].
Pathological study of the sac revealed an inner layer of endothelial cells, elastin tissues, and an outer layer of muscular tissue that resembled more a vessel than the classic angiomatous hamartoma seen in a typical arteriovenous fistula [28–30].
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Anatomic Variations
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Based upon the anatomical descriptions encountered in the reported cases and angiographic findings, de Souza e Silva and coworkers categorized three types of connections. Later, Ohara and coworkers added a fourth type.
Type I is an anomalous vessel connecting the posterior aspect of the proximal right pulmonary artery and draining into the left atrium with normal pulmonary venous connections.
Type II is a persistent and supernumerary branch of pulmonary artery that establishes abnormal connection with surrounding developing vascular structures—left atrium or pulmonary vein, or both, in the embryonic life. The right inferior pulmonary vein is absent. The lower lobe branch of the right pulmonary artery connection or fistulous tract joins the right inferior pulmonary vein and is aneurysmal proximal to its junction with the left atrium. Such communications between the pulmonary artery and the left atrium in type II may be the result of agenesis of the lobe of the lung and consequent absence of the pulmonary capillary bed.
In type III, all pulmonary veins drain into the aneurysmal pouch between the right pulmonary artery and left atrium.
In type IV, a very proximal pulmonary arteriovenous fistula joins the left atrium. The right-sided pulmonary veins join the fistulous tract and the left-sided pulmonary veins join the left atrium directly (Fig 1) [21, 31].
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Fig 1. Diagramatic representation of various types of right pulmonary artery to left atrium communications. Type I: Normal pulmonary venous pattern. Type II: Absent right inferior pulmonary vein with fistulous connection at the normal site of its ostium. Type III: All pulmonary veins connected to the aneurysmal pouch. Type IV: Right inferior pulmonary vein replaced by three small veins connected to the aneurysmal pouch. The left-sided pulmonary veins joined the left atrium directly. (LA = left atrium; LLPV = left lower pulmonary vein; LPA = left pulmonary artery; LUPV = left upper pulmonary vein; LV = left ventricle; P.T. = pulmonary trunk; RA = right atrium; RLPV = right lower pulmonary vein; RMPV = right middle pulmonary vein; RPA = right pulmonary artery; RUPV = right upper pulmonary vein; RV = right ventricle.)
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These fistulas vary in size from very small without any clinical significance, to very large with congestive cardiac failure. Usually, the opening of the fistula is located at the posterior side of the right pulmonary artery. The opening of the fistula at the side of the left atrium can be located high or low in the atrium [9]. Only 3 cases of communication between left pulmonary artery and left atrium have been reported in the world literature [5, 32, 33]. The frequent involvement of the right pulmonary artery as compared with the left is interesting but remains unexplained.
Associated Anomalies
This constant location with repetitive anatomy of pulmonary venous drainage indicates that this entity is a separate congenital cardiovascular disease, rather than part of the pulmonary arteriovenous malformations, which in 60% of cases are associated with hereditary hemorrhagic telangiectasia (Rendu-Osler-Weber syndrome) [34].
Authors reporting cases in the newborn period have made specific reference to the presence of a patent ductus arteriosus in 7 of 10 cases [5, 7–9, 11–13]. This additional lesion is probably one of the main factors leading to early death of these patients [5, 7–9, 11–13]. Additional cardiovascular lesions reported have been atrial septal defect, left pulmonary artery stenosis, pulmonary or aortic valve regurgitation and forehead hemangiomas [2, 3, 5, 7–9, 13, 18, 20, 21, 23, 26, 31, 32, 35]. The right lung anomalies were reported by the Lucas, Kroeker, Nanoyama, and Abe groups [18, 20, 30, 35]. The case described by Kroeker and coworkers [20] was type I. Other cases were type II. All the cases of type II except 1 case, which was not mentioned, included right lung anomalies [5]. Agenesis of the lower or middle lobe, right lung sequestration, and diverticulum of the right main bronchus were the various anomalies reported so far [2, 3, 5, 7–9, 18–23, 31, 35].
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Surgical Timing, Approach, and Results
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Careful analysis of the published series of right pulmonary artery to left atrium communication including the present patient population favors early surgical intervention (Table 1) [1–9, 11–13, 18–26, 31, 32]. The ultimate goal is to prevent chronic arterial hypoxemia, systemic thromboembolic complications, congestive cardiac failure, and shunt-derived pulmonary hypertension [1–9]. Severe heart failure in newborns must be treated as a surgical emergency [5, 7–9, 11–13]. Newborns who have mild cyanosis and no apparent heart failure, however, may respond to medical management; therefore, surgery can be postponed [7]. The reported mortality rate of those undergoing all types of surgical repair has been high (22%). The majority of the reported surgical mortality occurred with procedures performed between 1950 and 1979. Types II and III were predominantly associated with poor outcomes. More recently, the mortality has approached zero. Surgical mortality was most commonly attributed to pulmonary embolism, hemorrhage, and cardiac failure [1–9, 12, 13, 15, 17–26, 31–35].
The literature documents 3 isolated case reports of successful transcatheter closure of the fistula [11, 14, 36]. Slack and coworkers [11] have recently demonstrated the first case in which a direct right pulmonary artery to left atrium fistula was definitively treated with catheter-based coil (Gianturco coil) closure in a neonate. The coil diameter was selected so that it was at least twice the diameter of the narrowed portion and also that the diameter did not exceed the size of the maximum diameter of the fistula at its origin from the right pulmonary artery. Coil length was selected so that the deployed coil would not occupy more than half the distance from the narrowed point back to the right pulmonary artery origin. This was believed to be important in order to minimize the potential for coil compromise of right pulmonary artery flow [11].
Subsequently, Zanchetta and coworkers [14] reported the first successful complete closure of a large right pulmonary artery to left atrium fistula in an adult patient using the Amplatzer duct occluder device. These authors addressed the possible limitations of various commercially available catheter-based coil closure systems to occlude large adult right pulmonary artery to left atrium fistulas. A detachable Gianturco coil was not used because of two reasons: (1) multiple coils might be required for complete occlusion of such large fistulous connection, and (2) it might be technically difficult to not jeopardize distal viable side branches of the right pulmonary artery and to avoid pulmonary infarction. The Bioptome-assisted coil delivery system was not used because navigating the Bioptome in the correct position through the pulmonary vessels seemed to be cumbersome. The Gianturco-Grifka vascular occlusion device was considered inappropriate because of the large size of the fistula compared with the maximum size available (9 mm) of such a device [14].
From a surgical view point, it is indeed necessary to accurately localize the lesion. Depending on the anatomical locations, the lesions have been approached through median sternotomy, right posterolateral thoracotomy, or left posterolateral thoracotomy [1–9, 11–18, 27, 28, 37–45]. In 29 patients, a right thoracotomy was performed, whereas 19 patients underwent median sternotomy. The lesion in 1 patient was unapproachable through right anterolateral thoracotomy, and a median sternotomy was performed to ligate the same. Five patients were managed medically, and the approach was unspecified in 3 patients. Left thoracotomy was performed in 3 cases of left pulmonary artery to left atrium fistula [1–9, 13–15, 27, 28, 37–45]. In the literature, 52.5% of the patients underwent ligation alone, 10.1% had ligation and division, 8.4% had excision, 10.1% had intracardiac repair, and 1.7% underwent pneumonectomy. The cases requiring intracardiac repair were the ones with difficult dissection, or short and wide fistula. Extracorporeal circulation was employed in 31 patients and in patients with associated cardiac anomalies (Table 1) [1–9, 13–15, 27, 28, 37–45]. Taking into account the variations in anatomy in the published literature and classifying them based on the pulmonary venous connection, an individualized management algorithm appears justifiable (Table 1).
Simple ligation or division through median sternotomy is especially suitable for type I fistulas. A right or left posterolateral thoracotomy may be the preferred approach for type II fistulas. The type III and type IV fistulas with involvement of pulmonary veins may require intracardiac repair with the use of extracorporeal circulation to pinpoint the opening of the anomalous vessel and the pulmonary vein from the left atrium [2, 3, 23, 31, 35].
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Conclusions
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In conclusion, the clinical diagnosis of right pulmonary artery to left atrium communications is difficult. Doppler echocardiography and angiographic investigation are mandatory to provide necessary diagnostic information and define the anatomy. The surgical treatment should be individualized and performed as soon as the diagnosis is made.
Simple ligation or division through median sternotomy appears suitable for type I fistulas. A right or left posterolateral thoracotomy may be the preferred approach for type II fistulas. Use of extracorporeal circulation may be better suited for type III and type IV fistulas, in instances where the dissection of the anomalous vessel is technically difficult, and in cases requiring intricate intracardiac repair. Despite a limited experience, catheter-based coil closure or an Amplatzer duct occluder may be considered in selected instances.
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Abstract
Introduction
