Eur J Cardiothorac Surg 2002;22:465-467
© 2002 Elsevier Science NL
Acquired pulmonary vein obstruction after open-heart surgery
Gian Paolo Ussia,
Maurizio Marasini*,
Lucio Zannini,
Giacomo Pongiglione
Laboratory of Invasive Cardiology, Pediatric Cardiology and Cardiac Surgery Department, Giannina Gaslini Institute, Children's Hospital, Largo Girolamo Gaslini, 5, 16147 Genova, Italy
Received 2 January 2002;
received in revised form 16 April 2002;
accepted 10 May 2002.
* Corresponding author. Tel.: +39-010-5636250; fax: +39-010-377-6590
e-mail: mauriziomarasini{at}ospedale-gaslini.ge.it
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Abstract
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Acquired pulmonary vein obstruction is an extremely rare complication after open-heart surgery not including pulmonary vein procedures. We report on three cases of this unusual complication presenting peculiar angiographic findings in order to understand its possible etiology.
Key Words: Pulmonary vein • Stenosis • Pneumonia • Wedge angiography
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1. Introduction
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Acquired obstruction of pulmonary veins (PV) has been extensively reported after surgical correction of anomalous pulmonary venous drainage and after atrial switch operation for transposition of the great arteries [1,2]. However, this complication is extremely rare in other types of open-heart surgery. We describe the unusual finding of acquired left PV obstruction in three patients who underwent cardiac surgery for different congenital heart diseases (CHD) without any surgical procedure on PV or left atrial (LA) structure.
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2. Case reports
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2.1. Case 1
An 11-day-old male infant underwent modified Norwood operation for tricuspid atresia with great arteries malposition, and type B aortic arch interruption. The early postoperative course was complicated by low cardiac output and deep desaturation requiring delayed sternum closure (25 days). Six months later the child was doing well, but chest X-ray showed left lung congestion compared to the normal appearance of lung perfusion at the time of hospital discharge. Angiography showed a small left pulmonary artery (LPA), with poor antegrade flow and a wide aortic to LPA collateral circulation which was considered at that time the cause of left lung congestion and of unopacified left PV at LPA injection. Bidirectional cavo-pulmonary anastomosis and proximal LPA enlargement were then performed. Six months later LPA was found to be hypoplastic with only retrograde flow into the right pulmonary artery (RPA) (Fig. 1A)
; balloon occluded angiography confirmed the complete occlusion of left PV (Fig. 1B). After 32 months of follow-up the patient is in a stable clinical condition.
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Fig. 1. (a) LPA angiography through the superior vena cava failed to show lung perfusion with retrograde right pulmonary injection. (b) Balloon occluded LPA angiography confirmed a complete occlusion of left PV (arrow).
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2.2. Case 2
A 1-month-old male baby underwent open-heart atrioseptectomy and pulmonary artery banding for double outlet right ventricle with mitral valve stenosis and left ventricular hypoplasia. Seven months later, pulmonary banding had distally migrated, determining severe RPA and mild LPA stenosis. Left PV were normal at angiography (Fig. 2A)
whereas the inferior vena cava (IVC) was found distally occluded with a collateral circulation via the azygous vein to the superior vena cava. The patient underwent fenestrated intracardiac total cavo-pulmonary connection and RPA arterioplasty extended toward LPA. The early postoperative period was complicated by persistent low O2 saturation and the need for prolonged thoracic drainage (15 days). The child was discharged at home 1 month later with 86% systemic saturation at rest and normal lung perfusion on chest X-ray. Cardiac catheterization was scheduled 6 months later and at that time chest X-ray showed an inferior lobe interstitial edema of the left lung. At selective balloon occluded LPA injection, the inferior PV was found to be completely obstructed (Fig. 2B) whereas aortic angiogram showed a wide collateral circulation from the left subclavian artery to LPA and back to RPA. After 30 months the baby is in a stable condition.
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Fig. 2. (a) 45° Left anterior oblique (LAO) angiography of LPA before surgery clearly showed patent left PVs (arrow). (b) LPA wedge angiography 6 months after surgery showed complete left PV obstruction (arrow).
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2.3. Case 3
A 1-day-old male baby admitted with diagnosis of transposition of the great arteries and aortic coarctation underwent balloon atrial septostomy and angiography performed at that time showed normal PV return. Arterial switch operation with coarctation repair was performed 8 days later. The early postoperative period was complicated by low cardiac output requiring delayed sternum closure (6 days) and prolonged thoracic drainage (21 days). The child was discharged home at 2 months of age in a stable condition and with normal bilateral lung perfusion on chest X-ray. Routine cardiac catheterization was scheduled after 1 year. At that time chest X-ray showed marked left lung congestion. Angiography showed multiple RPA stenosis, small LPA with retrograde flow into RPA, complete obstruction of left PV return and aorta-LPA collaterals. After 1 year of follow-up the baby is in a stable clinical condition.
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3. Discussion
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Congenital PV stenosis or atresia usually involves two or more veins of both lungs and become clinically evident in the first months of life with severe pulmonary hypertension, recurrent pneumonia, and failure to thrive. It is a dramatic clinical picture with a dismal prognosis even in the case of early diagnosis. Any attempts of therapy have given poor results [3].
On the contrary patients affected by acquired unilateral PV stenosis or obstruction become symptomatic later in life, and their clinical picture is non-specific and characterized by a more favorable course especially for the absence of contralateral lung hypertension at mid-term follow-up [3,7]. This condition is rare after an open-heart procedure unrelated to PV surgery or an atrial switch operation. Six cases have been reported to date and in all but one PV atresia was considered congenital and unidentified before surgery [3–6]. In our series, the patients had different CHD and congenital PV atresia was ruled out at angiography in two of them. Preoperative cross-sectional echocardiography visualization of left PV and the absence of left lung congestion on chest X-ray before surgery supported the hypothesis of acquired etiology even in the remaining patient.
Surgical procedures were far from the left PV and traumatic maneuvers on these vessels were avoided during the operation, with the exception of LA vent and postoperative LA line for pressure monitoring, both placed through the right PV. The early postoperative period was complicated, requiring delayed sternal closure and long mechanical ventilation or prolonged thoracic drainage in all patients. At discharge lung perfusion was apparently normal on chest X-ray whereas left pulmonary congestion was found late at follow-up. The common postoperative angiographic findings at left PV obstruction diagnosis were: hypoplastic LPA, inverted blood flow from LPA to RPA and PV obstruction confirmed by balloon occluded or wedge LPA angiography. A fine aortic-LPA collateral circulation with high LPA O2 saturation was also observed in all our patients. We speculate that once the left PV becomes atretic, the lung involved will not be supplied by the LPA, and aorto-pulmonary collateral vessels will develop. We think that the inverted blood flow observed in the LPA is the epiphenomenon of this new hemodynamic condition. Similarly, the small diameter LPA found at follow-up may be a consequence of left PV obstruction because LPA was normal before surgery in two cases and only slightly stenotic in one case.
We cannot exclude that intraoperative LA venting or long standing LA line could cause endothelial damage and potential lesion to left PV, particularly in a neonatal heart. However, direct damage of the PV should be ruled out, based on the normal bilateral appearance of lung perfusion at chest X-ray at the time of discharge from hospital.
Once direct traumatism of the left PV was excluded the only common feature to the three patients was a complicated postoperative course requiring delayed sternal closure and/or prolonged thoracic drainage. Thus, we can speculate that these conditions could have caused mediastinum fibrosis and progressive PV obstruction.
In otherwise unexplained unilateral pulmonary congestion after cardiac surgery, the PV should be carefully investigated with catheterization mainly in those patients with complicated postoperative course.
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References
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Abstract
1. Introduction
