Pulmonary vein obstruction after primary sutureless pericardial repair of a total anomalous pulmonary venous connection

Pulmonary vein obstruction after primary sutureless pericardial repair of a total anomalous... Abstract Total anomalous pulmonary venous connection is a rare congenital heart disease. The development of pulmonary venous obstruction is one of the major risk factors for poor outcomes after surgical repair. Sutureless pericardial repair for a total anomalous pulmonary venous connection was introduced to decrease the risk of pulmonary venous obstruction after surgical repair, and favourable outcomes have been reported. Herein, we report the case of an infant with a total anomalous pulmonary venous connection who developed pulmonary venous obstruction after primary sutureless pericardial repair. Total anomalous pulmonary venous connection , Pulmonary vein obstruction , Sutureless pericardial repair INTRODUCTION The development of pulmonary venous obstruction (PVO) is one of the major risk factors for postoperative hospital mortality after surgical repair of a total anomalous pulmonary venous connection [1, 2]. PVO remains an issue, and primary sutureless pericardial repair has been introduced to avoid the risk of developing PVO, and favourable outcomes have been reported with this approach. CASE REPORT A male infant was diagnosed with a total anomalous pulmonary venous connection (III), and surgical repair was performed soon after birth. The pulmonary vein (PV) chamber was anastomosed to the left atrium with sutureless pericardial repair (Fig. 1A). The longitudinal incision was localized to the PV chamber and did not extend beyond each PV branch to avoid intimal hyperplasia of PV branches possibly caused by mechanical stimuli. The diameter of PV branches appeared adequate on gross examination. After the surgery, a computed tomography (CT) scan revealed an anatomical anastomosis site measuring 9.5 mm in diameter. The patient had no symptoms possibly caused by PVO. Four years after primary sutureless pericardial repair was performed, manometric examination revealed a pressure gradient of 10 mmHg between the common PV chamber and the left atrium and a CT scan revealed that the diameter of the orifice was 7.6 mm, which was lower than the diameter measured soon after the primary repair (Fig. 1B and C). Surgical intervention under cardiopulmonary bypass for PVO was indicated and performed at 4 years and 4 months of age. Through a transatrial septal incision, the projecting tissue between the common PV chamber and left atrium (Fig. 2A), which was revealed to be a hypertrophied PV wall on pathological examination (Fig. 2B), was surgically resected. The postoperative echocardiography revealed that the estimated pressure gradient through the anastomosis site decreased to 1 mmHg, and the diameter of the site was 11 mm. Figure 1: View largeDownload slide (A) A drawing of primary sutureless pericardial repair. A dotted oval line shows the suture line on the pericardium. A longitudinal incision line was placed on the common pulmonary vein chamber. (B and C) The diameter of an orifice between the common pulmonary vein chamber and left atrium had decreased from 9.5 mm to 7.6 mm in the computed tomographic image. (B) An image immediately after the primary repair, (C) An image 4 years after the primary repair. Black arrow indicates left atrium, and red arrow indicates the common pulmonary vein chamber. Figure 1: View largeDownload slide (A) A drawing of primary sutureless pericardial repair. A dotted oval line shows the suture line on the pericardium. A longitudinal incision line was placed on the common pulmonary vein chamber. (B and C) The diameter of an orifice between the common pulmonary vein chamber and left atrium had decreased from 9.5 mm to 7.6 mm in the computed tomographic image. (B) An image immediately after the primary repair, (C) An image 4 years after the primary repair. Black arrow indicates left atrium, and red arrow indicates the common pulmonary vein chamber. Figure 2: View largeDownload slide (A) Intraoperative view through the atrial septum which was incised open. Black arrow shows the communication between the common pulmonary vein chamber and left atrium, and black arrowhead shows the blacked area of protruded pulmonary vein wall that caused pulmonary vein obstruction. After the resection of this tissue, 4 pulmonary vein orifices were observed. (B) Pathological examination of resected tissue revealed hypertrophied pulmonary vein wall. IVC: inferior vena cava; RA: right atrium; SVC: superior vena cava. Figure 2: View largeDownload slide (A) Intraoperative view through the atrial septum which was incised open. Black arrow shows the communication between the common pulmonary vein chamber and left atrium, and black arrowhead shows the blacked area of protruded pulmonary vein wall that caused pulmonary vein obstruction. After the resection of this tissue, 4 pulmonary vein orifices were observed. (B) Pathological examination of resected tissue revealed hypertrophied pulmonary vein wall. IVC: inferior vena cava; RA: right atrium; SVC: superior vena cava. DISCUSSION To treat postoperative PVO, which has been reported to be a risk factor for poor outcome, sutureless pericardial repair was introduced [3], and thus, sutureless pericardial repair has been indicated as the primary repair approach for a total anomalous pulmonary venous connection and shown to be associated with a low incidence of postoperative PVO [4]. The theoretical advantage of sutureless pericardial repair is the lateralization of the suture line away from the confluence edge of the PVs, minimizing mechanical stimuli associated with the development of obstruction. However, the lateralization of the suture line allows the pericardial edge, common PV chamber wall or left atrial wall to expand inside the suture line. In our case, sutureless pericardial repair was used as the primary repair. The longitudinal incision was localized to the common PV chamber and did not extend beyond each PV branch because we confirmed the adequate diameter of each PV, and there was no attachment of the edge of the PV chamber to the edge of the pericardium. However, the projection of tissue between the left atrium and PV chamber with a pressure gradient of 10 mmHg was observed in 4 years, and the orifice of the anastomosis site had decreased. The projecting tissue was surgically resected, and PVO was released. This projecting tissue was indicated as a hypertrophied PV wall. Because the diameter of each PV branch was conserved, PVO did not extend to each branch of the PV. Making incision on each PV branch may not be required to avoid mechanical stimuli when the diameter of PVs is confirmed to be sufficient; nonetheless, it is essential to anchor the edge of PV chamber wall and left atrial wall inside the suture line to maintain the orifice of the anastomosed site and to prevent the protrusion of the pericardial or PV wall as observed in our case. Conflict of interest: none declared. REFERENCES 1 Hlavacek AM, Shirali GS, Anderson RH. Pulmonary Venous Abnormalities. In: Anderson BR, Baker EJ, Redington A, Rigby ML, Penny D, Wernovsky G (eds). Paediatric Cardiology , 3rd edn, Vol. 24. Philadelphia, PA: Churchill Livingstone, Elsevier, 2010, 497– 522. 2 Lewis FJ, Varco RL, Taufic M, Niazi SA. Direct vision repair of triatrial heart and total anomalous pulmonary venous drainage. Surg Gynecol Obstet  1956; 102: 713– 20. Google Scholar PubMed  3 Najm HK, Caldarone CA, Smallhorn J, Coles JG. Sutureless technique for the relief of pulmonary vein stenosis with the use of in situ pericardium. J Thorac Cardiovasc Surg  1998; 115: 468– 70. Google Scholar CrossRef Search ADS PubMed  4 Yanagawa B, Alghamdi AA, Dragulescu A, Viola N, Al-Radi OO, Mertens LL et al.   Primary sutureless repair for simple total anomalous pulmonary venous connection: midterm result in a single institution. J Thorac Cardiovasc Surge  2011; 141: 1346– 54. Google Scholar CrossRef Search ADS   © The Author(s) 2018. Published by Oxford University Press on behalf of the European Association for Cardio-Thoracic Surgery. All rights reserved. This article is published and distributed under the terms of the Oxford University Press, Standard Journals Publication Model (https://academic.oup.com/journals/pages/about_us/legal/notices) http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Interactive CardioVascular and Thoracic Surgery Oxford University Press

Pulmonary vein obstruction after primary sutureless pericardial repair of a total anomalous pulmonary venous connection

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Oxford University Press
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© The Author(s) 2018. Published by Oxford University Press on behalf of the European Association for Cardio-Thoracic Surgery. All rights reserved.
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1569-9293
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1569-9285
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10.1093/icvts/ivy096
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Abstract

Abstract Total anomalous pulmonary venous connection is a rare congenital heart disease. The development of pulmonary venous obstruction is one of the major risk factors for poor outcomes after surgical repair. Sutureless pericardial repair for a total anomalous pulmonary venous connection was introduced to decrease the risk of pulmonary venous obstruction after surgical repair, and favourable outcomes have been reported. Herein, we report the case of an infant with a total anomalous pulmonary venous connection who developed pulmonary venous obstruction after primary sutureless pericardial repair. Total anomalous pulmonary venous connection , Pulmonary vein obstruction , Sutureless pericardial repair INTRODUCTION The development of pulmonary venous obstruction (PVO) is one of the major risk factors for postoperative hospital mortality after surgical repair of a total anomalous pulmonary venous connection [1, 2]. PVO remains an issue, and primary sutureless pericardial repair has been introduced to avoid the risk of developing PVO, and favourable outcomes have been reported with this approach. CASE REPORT A male infant was diagnosed with a total anomalous pulmonary venous connection (III), and surgical repair was performed soon after birth. The pulmonary vein (PV) chamber was anastomosed to the left atrium with sutureless pericardial repair (Fig. 1A). The longitudinal incision was localized to the PV chamber and did not extend beyond each PV branch to avoid intimal hyperplasia of PV branches possibly caused by mechanical stimuli. The diameter of PV branches appeared adequate on gross examination. After the surgery, a computed tomography (CT) scan revealed an anatomical anastomosis site measuring 9.5 mm in diameter. The patient had no symptoms possibly caused by PVO. Four years after primary sutureless pericardial repair was performed, manometric examination revealed a pressure gradient of 10 mmHg between the common PV chamber and the left atrium and a CT scan revealed that the diameter of the orifice was 7.6 mm, which was lower than the diameter measured soon after the primary repair (Fig. 1B and C). Surgical intervention under cardiopulmonary bypass for PVO was indicated and performed at 4 years and 4 months of age. Through a transatrial septal incision, the projecting tissue between the common PV chamber and left atrium (Fig. 2A), which was revealed to be a hypertrophied PV wall on pathological examination (Fig. 2B), was surgically resected. The postoperative echocardiography revealed that the estimated pressure gradient through the anastomosis site decreased to 1 mmHg, and the diameter of the site was 11 mm. Figure 1: View largeDownload slide (A) A drawing of primary sutureless pericardial repair. A dotted oval line shows the suture line on the pericardium. A longitudinal incision line was placed on the common pulmonary vein chamber. (B and C) The diameter of an orifice between the common pulmonary vein chamber and left atrium had decreased from 9.5 mm to 7.6 mm in the computed tomographic image. (B) An image immediately after the primary repair, (C) An image 4 years after the primary repair. Black arrow indicates left atrium, and red arrow indicates the common pulmonary vein chamber. Figure 1: View largeDownload slide (A) A drawing of primary sutureless pericardial repair. A dotted oval line shows the suture line on the pericardium. A longitudinal incision line was placed on the common pulmonary vein chamber. (B and C) The diameter of an orifice between the common pulmonary vein chamber and left atrium had decreased from 9.5 mm to 7.6 mm in the computed tomographic image. (B) An image immediately after the primary repair, (C) An image 4 years after the primary repair. Black arrow indicates left atrium, and red arrow indicates the common pulmonary vein chamber. Figure 2: View largeDownload slide (A) Intraoperative view through the atrial septum which was incised open. Black arrow shows the communication between the common pulmonary vein chamber and left atrium, and black arrowhead shows the blacked area of protruded pulmonary vein wall that caused pulmonary vein obstruction. After the resection of this tissue, 4 pulmonary vein orifices were observed. (B) Pathological examination of resected tissue revealed hypertrophied pulmonary vein wall. IVC: inferior vena cava; RA: right atrium; SVC: superior vena cava. Figure 2: View largeDownload slide (A) Intraoperative view through the atrial septum which was incised open. Black arrow shows the communication between the common pulmonary vein chamber and left atrium, and black arrowhead shows the blacked area of protruded pulmonary vein wall that caused pulmonary vein obstruction. After the resection of this tissue, 4 pulmonary vein orifices were observed. (B) Pathological examination of resected tissue revealed hypertrophied pulmonary vein wall. IVC: inferior vena cava; RA: right atrium; SVC: superior vena cava. DISCUSSION To treat postoperative PVO, which has been reported to be a risk factor for poor outcome, sutureless pericardial repair was introduced [3], and thus, sutureless pericardial repair has been indicated as the primary repair approach for a total anomalous pulmonary venous connection and shown to be associated with a low incidence of postoperative PVO [4]. The theoretical advantage of sutureless pericardial repair is the lateralization of the suture line away from the confluence edge of the PVs, minimizing mechanical stimuli associated with the development of obstruction. However, the lateralization of the suture line allows the pericardial edge, common PV chamber wall or left atrial wall to expand inside the suture line. In our case, sutureless pericardial repair was used as the primary repair. The longitudinal incision was localized to the common PV chamber and did not extend beyond each PV branch because we confirmed the adequate diameter of each PV, and there was no attachment of the edge of the PV chamber to the edge of the pericardium. However, the projection of tissue between the left atrium and PV chamber with a pressure gradient of 10 mmHg was observed in 4 years, and the orifice of the anastomosis site had decreased. The projecting tissue was surgically resected, and PVO was released. This projecting tissue was indicated as a hypertrophied PV wall. Because the diameter of each PV branch was conserved, PVO did not extend to each branch of the PV. Making incision on each PV branch may not be required to avoid mechanical stimuli when the diameter of PVs is confirmed to be sufficient; nonetheless, it is essential to anchor the edge of PV chamber wall and left atrial wall inside the suture line to maintain the orifice of the anastomosed site and to prevent the protrusion of the pericardial or PV wall as observed in our case. Conflict of interest: none declared. REFERENCES 1 Hlavacek AM, Shirali GS, Anderson RH. Pulmonary Venous Abnormalities. In: Anderson BR, Baker EJ, Redington A, Rigby ML, Penny D, Wernovsky G (eds). Paediatric Cardiology , 3rd edn, Vol. 24. Philadelphia, PA: Churchill Livingstone, Elsevier, 2010, 497– 522. 2 Lewis FJ, Varco RL, Taufic M, Niazi SA. Direct vision repair of triatrial heart and total anomalous pulmonary venous drainage. Surg Gynecol Obstet  1956; 102: 713– 20. Google Scholar PubMed  3 Najm HK, Caldarone CA, Smallhorn J, Coles JG. Sutureless technique for the relief of pulmonary vein stenosis with the use of in situ pericardium. J Thorac Cardiovasc Surg  1998; 115: 468– 70. Google Scholar CrossRef Search ADS PubMed  4 Yanagawa B, Alghamdi AA, Dragulescu A, Viola N, Al-Radi OO, Mertens LL et al.   Primary sutureless repair for simple total anomalous pulmonary venous connection: midterm result in a single institution. J Thorac Cardiovasc Surge  2011; 141: 1346– 54. Google Scholar CrossRef Search ADS   © The Author(s) 2018. Published by Oxford University Press on behalf of the European Association for Cardio-Thoracic Surgery. All rights reserved. This article is published and distributed under the terms of the Oxford University Press, Standard Journals Publication Model (https://academic.oup.com/journals/pages/about_us/legal/notices)

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Interactive CardioVascular and Thoracic SurgeryOxford University Press

Published: Apr 2, 2018

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