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J Thorac Cardiovasc Surg 1995;110:1563-1565
© 1995 Mosby, Inc.

BRIEF COMMUNICATIONS

THROMBOSIS OF THE PULMONARY ARTERY STUMP AFTER DISTAL LIGATION

New Haven, Conn.

Accepted for publication May 31, 1995. Patients who have undergone a Fontan operation are at risk for the development of thrombi in a variety of locations, including the caval system, the systemic venous atrium, the distal pulmonary arteries, and the systemic ventricle. ^1-3 Recently, in several patients born with univentricular heart and antegrade pulmonary flow, we observed thrombi within the pulmonary artery stump after distal ligation of the main pulmonary artery. Thrombosis has not previously been documented in this location. This finding has implications regarding the optimal medical and surgical management for these patients.

Case 1.

A male infant was born at term with transposition of the great arteries, double aortic arch, and absent interventricular septum, precluding biventricular repair. He initially underwent ligation of the ductus arteriosus, division of the left aortic arch, and banding of the pulmonary artery. At the age of 6 months, he underwent a bidirectional Glenn anastomosis with distal ligation of the main pulmonary artery to preclude antegrade flow. He subsequently did well. At the age of 26 months, he underwent elective cardiac catheterization in preparation for a total cavopulmonary connection. He was in sinus rhythm, with the hemoglobin value and platelet count being within normal limits.

As is our current practice for patients undergoing staged procedures toward the Fontan operation, we performed transesophageal echocardiography immediately before catheterization. This demonstrated a 1 cm sessile thrombus within the stump of the ligated pulmonary artery, immediately distal to the pulmonic valve. The pulmonic valve opened partially, and color flow Doppler echocardiography showed flow into the pulmonary artery, with regurgitation also seen. Ventricular function was excellent, with no other thrombi found within the heart or vascular structures. Angiography demonstrated that contrast material entered the pulmonary artery, outlining the thrombus. Clearance of contrast material from this site was delayed.

Case 2.

A male infant was born at term with double-inlet left ventricle, L-malposition of the great arteries, and mild pulmonic stenosis. At the age of 2 months, he underwent banding of the pulmonary artery, followed at the age of 9 months by a bidirectional Glenn anastomosis with ligation of the distal main pulmonary artery. When he was 15 months old, he underwent cardiac catheterization before undergoing the total cavopulmonary connection. He was in sinus rhythm, with a mildly elevated hemoglobin level (15.3 gm/dl) and a platelet count within the normal range.

Transesophageal echocardiography demonstrated a very large thrombus within the main pulmonary artery stump, occupying its entire volume (Fig. 1). Color flow Doppler echocardiography demonstrated antegrade flow into the pulmonary artery with regurgitation. The ventricular function was good, with no other thrombi noted. Angiography showed contrast material entering the pulmonary artery and outlining the thrombus, with extremely delayed clearance of contrast material from the pulmonary artery stump.

View larger version (62K): [in this window] [in a new window]   Fig. 1. Transesophageal echocardiogram showing crosssection of the main pulmonary artery (MPA), just above the level of the valve, with thrombus filling the artery.

A male infant was born at term with tricuspid atresia, ventricular septal defect, and normally related great arteries. A classic right Blalock-Taussig shunt was performed at the age of 2 weeks. Postoperatively, thrombosis of the sagittal sinus developed. Hemoglobin, platelet count, protein C and S, antithrombin III, and plasminogen levels were all within normal limits. He recovered from these events and did well. At the age of 10 months, he underwent a bidirectional Glenn anastomosis, takedown of the Blalock-Taussig shunt, and ligation of the distal main pulmonary artery. When he was 18 months old, cardiac catheterization was performed in preparation for the total cavopulmonary connection. The patient was in normal sinus rhythm, with elevated hemoglobin values (16.5 gm/dl) and a platelet count within the normal range.

Transesophageal echocardiography demonstrated a very large thrombus within the main pulmonary artery, just distal to the pulmonic valve (Fig. 2). The thrombus filled virtually the entire artery, interfering with pulmonic valve motion. Valvular regurgitation was noted. Ventricular function was good, with no other thrombi noted. Angiography demonstrated faint opacification of the pulmonary artery stump with delayed clearance of contrast material.

View larger version (98K): [in this window] [in a new window]   Fig. 2. Transesophageal echocardiogram showing the heart in short axis at the level of the aorta. A large thrombus is seen just above the level of the pulmonic valve, occluding the artery. RVOT, Right ventricular outflow tract; CLOT, thrombus; AO, aorta; MPA, main pulmonary artery; LV, left ventricle.

These three patients had certain anatomic similarities, that is, a univentricular heart with antegrade flow into the pulmonary artery. As part of the surgical staging toward a Fontan procedure, they underwent a bidirectional Glenn anastomosis, with ligation of the distal main pulmonary artery to exclude antegrade pulmonic flow. This ligation creates a blind stump, which appears to be a potential nidus for thrombus formation, presumably because of the presence of stagnant flow. Furthermore, if pulmonic regurgitation is present (as in these three patients) such a thrombus may gain access to the systemic ventricle with the potential for systemic embolic complications. Although our patients appear to have escaped such complications, these problems are certainly known to occur in patients having the Fontan operation. ^1-5

The occurrence of these thrombi in this location highlights the importance of aggressive evaluation of patients such as these and further suggests that prevention of thrombus should be a consideration when antegrade pulmonary blood flow is being curtailed. Although the optimal surgical technique for exclusion of the pulmonary artery from the systemic ventricle is not clearly defined, consideration should be given to patch closure or primary closure at the level of the pulmonic valve, rather than (or in addition to) ligation of the distal pulmonary artery.

Footnotes

From the Yale University School of Medicine, New Haven, Conn.

J THORAC CORDIOVASC SURG 1995;110:1563-5

References

1. Dajee H, Deutsch LS, Benson LM, Perloff JK, Laks H. Thrombolytic therapy for superior vena caval thrombosis following superior vena cava–pulmonary anastomosis. Ann Thorac Surg 1984;38:637-9.[Abstract]
   
2. Putnam JB, Lemmer JH, Rocchini AP, Bove EL. Embolectomy for acute pulmonary artery occlusion following Fontan procedure. Ann Thorac Surg 1988;45:335-6.[Abstract]
   
3. Okita Y, Miki S, Kusuhara K, et al. Massive systemic venous thrombosis after Fontan operation: report of a case. Thorac Cardiovasc Surg 1988;36:234-6.[Medline]
   
4. Mahony L, Nikaidoh H, Fixler DE. Thrombolytic treatment with streptokinase for late intraatrial thrombosis after modified Fontan procedure. Am J Cardiol 1988;62:343-4.[Medline]
   
5. Dobell ARC, Trusler GA, Smallhorn JF, Williams WG. Atrial thrombi after the Fontan operation. Ann Thorac Surg 1986;42:664-7.[Abstract]
   

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