Massive haemorrhage at resternotomy after stent implantation in ventriculo-pulmonary shunt after Norwood procedure
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a Department of Cardiothoracic Surgery, Erasmus MC, Dr Molewaterplein 40, 3015 GD Rotterdam, The Netherlands
b Department of Cardiothoracic Surgery, Catharina Hospital, Michelangelolaan 2, 5623 EJ Eindhoven, The Netherlands
c Department of Pediatric Cardiology, Erasmus MC, Dr Molewaterplein 40, 3015 GD Rotterdam, The Netherlands
Abstract
The ventriculo-pulmonary shunt in hypoplastic left heart syndrome has become an alternative to the arterio-pulmonary shunt. We present a patient with a severe stenosis in the ventriculo-pulmonary shunt at the proximal anastomosis, for which emergency balloon dilatation and stent implantation was performed. The location of the stent in the shunt, leaving only a thin compressed infundibular myocardium directly beneath the sternum, predisposed to massive haemorrhage at resternotomy for bidirectional Glenn procedure.
Key Words: Hypoplastic left heart syndrome; Ventriculo-pulmonary shunt; Balloon dilatation; Stent
1. Introduction
The three staged palliative surgical management for children with hypoplastic left heart syndrome (HLHS) is now widely accepted [1,2]. In this regard, the modified Norwood procedure using a ventriculo-pulmonary shunt (VP) produces a stable systemic circulation as well as an adequate pulmonary flow, resulting in better postoperative haemodynamics, better ventricular function and improved early survival [2–4].
The most frequent complication of the VP shunt is progressive obstruction of the shunt, particularly from three months on after surgery, resulting in a decrease of oxygen saturation and an increase in pressure gradient across the shunt [3,4]. Although, in addition, less optimal pulmonary artery development as compared to the arterio-pulmonary shunt was described [4], recent studies showed that the VP shunt is associated with considerably improved growth of the central pulmonary arteries [5,6]. This may, however, be associated with more severe stenoses of the pulmonary arteries at the site of the shunt insertion, resulting in important interstage morbidity.
2. Case report
A 12-day-old male infant, weighing 3100 g, who was antenatally diagnosed with HLHS, underwent a modified Norwood operation with a VP shunt. The procedure included aortic reconstruction with a pulmonary homograft patch, and a Gore-Tex 5 mm shunt (W.L. Gore & Associates, Inc, Flagstaff, Ariz) between a small right ventriculotomy in the distal infundibulum rightward from the pulmonary valve, out of the midline, and the right pulmonary artery. The procedure was performed without complications and the patient was sent home after 12 days.
At the age of 11 weeks, he was readmitted because of progressive cyanosis and a transcutaneous oxygen saturation of 55–70%. He now weighed 4460 g. Angiography showed an extreme narrowing at the origin of the shunt at the right ventricle. An F4 Judkins right coronary catheter (Cordis, Johnson & Johnson, Miami Lakes, FL) was positioned at the origin of the shunt for selective angiography. The stenosis was crossed with a stiff Ironman guide wire (Guidant, Santa Clara, CA). Emergency balloon dilatation was performed with a 2.5-mm Maverick (Boston Scientific Corp., Natick, MA) and a 4-mm Tyshak II (NuMed Inc, Hopkinton, NY) balloon. Because this resulted in only little improvement, a 6-mm Omnilink stent (Guidant) was placed with a good result (Fig. 1). Transcutaneous oxygen saturation increased to 86%. After stent insertion, the patient was treated with warfarin with a target international normalized ratio of 2.0–3.0.
At the age of 4 months the patient weighed 6100 g and transcutaneous oxygen saturation had slowly decreased to 65%. The X-ray showed no change in the stent position. It was located directly under the sternum (Fig. 2). A bidirectional Glenn procedure was scheduled. Despite anterior lifting of the sternum, there was massive blood loss at resternotomy, accompanied by severe haemodynamic instability. Arterial cannulation in the right femoral artery was performed. Sternotomy was completed and a venous cannula was put in the lesion. This turned out to be the lacerated stent, which had perforated the anterior wall of the infundibulum just proximal from the ventricular anastomosis of the VP shunt. The arterial cannula was switched to the aorta, the venous cannula was switched to the right atrium. Then we could safely proceed and remove the stent. The VP shunt was closed and the infundibular lesion could be closed by primary suturing. After removing the prosthetic material from the pulmonary artery, a superior cavo-pulmonary anastomosis was performed without further complications. A Gore-Tex surgical membrane was inserted beneath the sternum. The postoperative course was uneventful and the patient was released after 10 days.
3. Discussion
The VP shunt to reestablish pulmonary blood supply in stage I palliation for hypoplastic left heart syndrome was first described by Norwood and colleagues in 1981 [7]. In 1998, Sano again pointed at a VP shunt with a small nonvalved PTFE conduit, with good postoperative haemodynamics [3]. This shunt provides adequate pulmonary blood flow, a more stable balance between systemic and pulmonary circulation and reduces interstage mortality [8].
Sano described his technique with anastomosis to the distal stump of the main pulmonary artery, the shunt passing to the left of the neo-aorta [3]. Recently, McGuirk et al. described a modification in passing it to the right of the neo-aorta [2], as was done in our patient. This modification allows for a short and straight segment of prosthesis, a position rightward from the midline and complete removal of prosthetic material from the pulmonary artery at the Glenn operation.
When the VP shunt was first used there was some concern about pulmonary artery growth, essential for a safe and successful cavo-pulmonary connection at a later stage [3,4]. Recently, Rumball et al. described that, compared with the arterio-pulmonary shunt, the VP shunt stimulates better growth of the pulmonary arteries, but that the VP shunt was associated with more central pulmonary artery stenosis [5].
Severe stenosis of the VP shunt is a serious complication, causing a decrease in oxygen saturation and an increased pressure gradient over the shunt. These stenoses typically occur within the central pulmonary arteries at the site of the shunt insertion, most of the time within three months after the Norwood procedure [5]. The majority of these patients require intervention prior to further surgery in order to gain age and growth of the patient and to increase the size of the pulmonary arteries, needed for a safe and successful cavo-pulmonary connection. Opposite to these reports, in our case there was a shunt stenosis at the connection of the shunt with the right ventricle. Because of the sudden deterioration and decrease in oxygen saturation, and because we thought that it was too early to perform a bidirectional Glenn at this stage safely, we decided to dilate the shunt and to insert a stent. Initially the result was adequate.
In retrospect, the complicated resternotomy at the PCPC procedure was, despite of anterior lifting of the sternum, probably due to both the position of the shunt with the proximal anastomosis, just right from the midline and passing to the right of the neo-aorta, and the location of the stent in the shunt. Probably the infundibulum was pushed upward by the recoiling stent, causing it to lie directly underneath the sternum, leaving only a thin compressed ventricular wall directly beneath the sternum.
In conclusion, we would like the surgical audience to pay particular attention at resternotomy to retrosternal stents pushing cardiac structures to the sternum, in order to prevent damage to essential cardiac structures especially after the Norwood procedure with a VP shunt.
References
Bove EL, Lloyd TR. Staged reconstruction for hypoplastic left heart syndrome. Contemporary results. Ann Surg 1996; 224:387–395.
McGuirk SP, Griselli M, Stumper O, Rumball EM, Miller P, Dhillon R, de Giovanni JV, Wright JG, Barron DJ, Brawn WJ. Staged surgical management of hypoplastic left heart syndrome: a single institution 12-year experience. Heart Jun 6, 2005; doi:10.1136/hrt.2005.068684.
Sano S, Ishino K, Kawada M, Arai S, Kasahara S, Tomohiro A, Masuda Z, Takeuchi M, Ohtsuki S. Right ventricle-pulmonary artery shunt in first-stage palliation of hypoplastic left heart syndrome. J Thorac Cardiovasc Surg 2003; 126:504–509.
Sano S, Ishino K, Kado H, Shiokawa Y, Sakamoto K, Yokota M, Kawada M. Outcome of right ventricle-to-pulmonary artery shunt in first-stage palliation of hypoplastic left heart syndrome: a multi-institutional study. Ann Thorac Surg 2004; 78:1951–1958.
Rumball EM, McGuirk SP, Stümper O, Laker SJ, de Giovanni JV, Wright JG, Barron DJ, Brawn WJ. The RV-PA conduit stimulates better growth of the pulmonary arteries in hypoplastic left heart syndrome. Eur J Cardiothorac Surg 2005; 27:801–806.
Januszewska K, Kolcz J, Mroczek T, Procelewska M, Malec E. Right ventricle-to-pulmonary artery shunt and modified Blalock-Taussing shunt in preparation to hemi-Fontan procedure in children with hypoplastic left heart syndrome. Eur J Cardiothorac Surg 2005; 27:956–661.
Norwood WI, Lang P, Castaneda AR, Campbell DN. Experience with operation for hypoplastic left heart syndrome. J Thorac Cardiovasc Surg 1981; 82:511–519.
Pizarro C, Mroczek T, Malec E, Norwood WI. Right ventricle to pulmonary artery conduit reduces interim mortality after stage 1 Norwood for hypoplastic left heart syndrome. Ann Thorac Surg 2004; 78:1959–1964.(Suzanne Kats, Peter L. de)
b Department of Cardiothoracic Surgery, Catharina Hospital, Michelangelolaan 2, 5623 EJ Eindhoven, The Netherlands
c Department of Pediatric Cardiology, Erasmus MC, Dr Molewaterplein 40, 3015 GD Rotterdam, The Netherlands
Abstract
The ventriculo-pulmonary shunt in hypoplastic left heart syndrome has become an alternative to the arterio-pulmonary shunt. We present a patient with a severe stenosis in the ventriculo-pulmonary shunt at the proximal anastomosis, for which emergency balloon dilatation and stent implantation was performed. The location of the stent in the shunt, leaving only a thin compressed infundibular myocardium directly beneath the sternum, predisposed to massive haemorrhage at resternotomy for bidirectional Glenn procedure.
Key Words: Hypoplastic left heart syndrome; Ventriculo-pulmonary shunt; Balloon dilatation; Stent
1. Introduction
The three staged palliative surgical management for children with hypoplastic left heart syndrome (HLHS) is now widely accepted [1,2]. In this regard, the modified Norwood procedure using a ventriculo-pulmonary shunt (VP) produces a stable systemic circulation as well as an adequate pulmonary flow, resulting in better postoperative haemodynamics, better ventricular function and improved early survival [2–4].
The most frequent complication of the VP shunt is progressive obstruction of the shunt, particularly from three months on after surgery, resulting in a decrease of oxygen saturation and an increase in pressure gradient across the shunt [3,4]. Although, in addition, less optimal pulmonary artery development as compared to the arterio-pulmonary shunt was described [4], recent studies showed that the VP shunt is associated with considerably improved growth of the central pulmonary arteries [5,6]. This may, however, be associated with more severe stenoses of the pulmonary arteries at the site of the shunt insertion, resulting in important interstage morbidity.
2. Case report
A 12-day-old male infant, weighing 3100 g, who was antenatally diagnosed with HLHS, underwent a modified Norwood operation with a VP shunt. The procedure included aortic reconstruction with a pulmonary homograft patch, and a Gore-Tex 5 mm shunt (W.L. Gore & Associates, Inc, Flagstaff, Ariz) between a small right ventriculotomy in the distal infundibulum rightward from the pulmonary valve, out of the midline, and the right pulmonary artery. The procedure was performed without complications and the patient was sent home after 12 days.
At the age of 11 weeks, he was readmitted because of progressive cyanosis and a transcutaneous oxygen saturation of 55–70%. He now weighed 4460 g. Angiography showed an extreme narrowing at the origin of the shunt at the right ventricle. An F4 Judkins right coronary catheter (Cordis, Johnson & Johnson, Miami Lakes, FL) was positioned at the origin of the shunt for selective angiography. The stenosis was crossed with a stiff Ironman guide wire (Guidant, Santa Clara, CA). Emergency balloon dilatation was performed with a 2.5-mm Maverick (Boston Scientific Corp., Natick, MA) and a 4-mm Tyshak II (NuMed Inc, Hopkinton, NY) balloon. Because this resulted in only little improvement, a 6-mm Omnilink stent (Guidant) was placed with a good result (Fig. 1). Transcutaneous oxygen saturation increased to 86%. After stent insertion, the patient was treated with warfarin with a target international normalized ratio of 2.0–3.0.
At the age of 4 months the patient weighed 6100 g and transcutaneous oxygen saturation had slowly decreased to 65%. The X-ray showed no change in the stent position. It was located directly under the sternum (Fig. 2). A bidirectional Glenn procedure was scheduled. Despite anterior lifting of the sternum, there was massive blood loss at resternotomy, accompanied by severe haemodynamic instability. Arterial cannulation in the right femoral artery was performed. Sternotomy was completed and a venous cannula was put in the lesion. This turned out to be the lacerated stent, which had perforated the anterior wall of the infundibulum just proximal from the ventricular anastomosis of the VP shunt. The arterial cannula was switched to the aorta, the venous cannula was switched to the right atrium. Then we could safely proceed and remove the stent. The VP shunt was closed and the infundibular lesion could be closed by primary suturing. After removing the prosthetic material from the pulmonary artery, a superior cavo-pulmonary anastomosis was performed without further complications. A Gore-Tex surgical membrane was inserted beneath the sternum. The postoperative course was uneventful and the patient was released after 10 days.
3. Discussion
The VP shunt to reestablish pulmonary blood supply in stage I palliation for hypoplastic left heart syndrome was first described by Norwood and colleagues in 1981 [7]. In 1998, Sano again pointed at a VP shunt with a small nonvalved PTFE conduit, with good postoperative haemodynamics [3]. This shunt provides adequate pulmonary blood flow, a more stable balance between systemic and pulmonary circulation and reduces interstage mortality [8].
Sano described his technique with anastomosis to the distal stump of the main pulmonary artery, the shunt passing to the left of the neo-aorta [3]. Recently, McGuirk et al. described a modification in passing it to the right of the neo-aorta [2], as was done in our patient. This modification allows for a short and straight segment of prosthesis, a position rightward from the midline and complete removal of prosthetic material from the pulmonary artery at the Glenn operation.
When the VP shunt was first used there was some concern about pulmonary artery growth, essential for a safe and successful cavo-pulmonary connection at a later stage [3,4]. Recently, Rumball et al. described that, compared with the arterio-pulmonary shunt, the VP shunt stimulates better growth of the pulmonary arteries, but that the VP shunt was associated with more central pulmonary artery stenosis [5].
Severe stenosis of the VP shunt is a serious complication, causing a decrease in oxygen saturation and an increased pressure gradient over the shunt. These stenoses typically occur within the central pulmonary arteries at the site of the shunt insertion, most of the time within three months after the Norwood procedure [5]. The majority of these patients require intervention prior to further surgery in order to gain age and growth of the patient and to increase the size of the pulmonary arteries, needed for a safe and successful cavo-pulmonary connection. Opposite to these reports, in our case there was a shunt stenosis at the connection of the shunt with the right ventricle. Because of the sudden deterioration and decrease in oxygen saturation, and because we thought that it was too early to perform a bidirectional Glenn at this stage safely, we decided to dilate the shunt and to insert a stent. Initially the result was adequate.
In retrospect, the complicated resternotomy at the PCPC procedure was, despite of anterior lifting of the sternum, probably due to both the position of the shunt with the proximal anastomosis, just right from the midline and passing to the right of the neo-aorta, and the location of the stent in the shunt. Probably the infundibulum was pushed upward by the recoiling stent, causing it to lie directly underneath the sternum, leaving only a thin compressed ventricular wall directly beneath the sternum.
In conclusion, we would like the surgical audience to pay particular attention at resternotomy to retrosternal stents pushing cardiac structures to the sternum, in order to prevent damage to essential cardiac structures especially after the Norwood procedure with a VP shunt.
References
Bove EL, Lloyd TR. Staged reconstruction for hypoplastic left heart syndrome. Contemporary results. Ann Surg 1996; 224:387–395.
McGuirk SP, Griselli M, Stumper O, Rumball EM, Miller P, Dhillon R, de Giovanni JV, Wright JG, Barron DJ, Brawn WJ. Staged surgical management of hypoplastic left heart syndrome: a single institution 12-year experience. Heart Jun 6, 2005; doi:10.1136/hrt.2005.068684.
Sano S, Ishino K, Kawada M, Arai S, Kasahara S, Tomohiro A, Masuda Z, Takeuchi M, Ohtsuki S. Right ventricle-pulmonary artery shunt in first-stage palliation of hypoplastic left heart syndrome. J Thorac Cardiovasc Surg 2003; 126:504–509.
Sano S, Ishino K, Kado H, Shiokawa Y, Sakamoto K, Yokota M, Kawada M. Outcome of right ventricle-to-pulmonary artery shunt in first-stage palliation of hypoplastic left heart syndrome: a multi-institutional study. Ann Thorac Surg 2004; 78:1951–1958.
Rumball EM, McGuirk SP, Stümper O, Laker SJ, de Giovanni JV, Wright JG, Barron DJ, Brawn WJ. The RV-PA conduit stimulates better growth of the pulmonary arteries in hypoplastic left heart syndrome. Eur J Cardiothorac Surg 2005; 27:801–806.
Januszewska K, Kolcz J, Mroczek T, Procelewska M, Malec E. Right ventricle-to-pulmonary artery shunt and modified Blalock-Taussing shunt in preparation to hemi-Fontan procedure in children with hypoplastic left heart syndrome. Eur J Cardiothorac Surg 2005; 27:956–661.
Norwood WI, Lang P, Castaneda AR, Campbell DN. Experience with operation for hypoplastic left heart syndrome. J Thorac Cardiovasc Surg 1981; 82:511–519.
Pizarro C, Mroczek T, Malec E, Norwood WI. Right ventricle to pulmonary artery conduit reduces interim mortality after stage 1 Norwood for hypoplastic left heart syndrome. Ann Thorac Surg 2004; 78:1959–1964.(Suzanne Kats, Peter L. de)