Accepted Manuscript Retroesophageal Total Anomalous Pulmonary Venous Connection in a Newborn Lindsay A. Caldarone, Christopher A. Caldarone, Luca A. Vricella PII:
S0022-5223(14)00446-2
DOI:
10.1016/j.jtcvs.2014.04.022
Reference:
YMTC 8561
To appear in:
The Journal of Thoracic and Cardiovascular Surgery
Received Date: 8 April 2014 Accepted Date: 11 April 2014
Please cite this article as: Caldarone LA, Caldarone CA, Vricella LA, Retroesophageal Total Anomalous Pulmonary Venous Connection in a Newborn, The Journal of Thoracic and Cardiovascular Surgery (2014), doi: 10.1016/j.jtcvs.2014.04.022. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Retroesophageal Total Anomalous Pulmonary Venous Connection in a Newborn
RI PT
Lindsay A. Caldarone1, Christopher A. Caldarone2, Luca A. Vricella1
Division of Cardiac Surgery, Johns Hopkins University School of Medicine, Baltimore, USA1
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EP
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SC
The Hospital for Sick Children, University of Toronto, Toronto, Canada2
Correspondence:
Luca A. Vricella, MD Director, Pediatric Cardiac Surgery and Heart Transplantation Johns Hopkins University, Baltimore, Maryland 1800 Orleans Street, Zayed 7107G Baltimore, MD 21287, USA Tel. (443) 287 - 1262 Fax (410) 955 - 3809 e-mail: [email protected]
WORD COUNT: 588
ACCEPTED MANUSCRIPT
Case Report
RI PT
A 2-month old, 4.3 Kg full-term infant with no prior cardiac diagnosis was found cyanotic and listless and emergently transported to our Institution, where a transthoracic echocardiogram revealed non-obstructed supracardiac total anomalous pulmonary venous connection (TAPVC) with a vertical vein coursing posterior to the left pulmonary artery and draining into the left brachiocephalic vein. The patient was brought to the operating room with the expectation that routine repair of supracardiac TAPVC would be performed.
Comment
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During surgery, however, the pulmonary venous confluence could not be identified through the posterior pericardium. Following cardioplegic and circulatory arrest at profound hypothermia, we attempted localization of the confluence and proceeded to even transect the inferior vena cava with the hope of finding the confluence once the heart could be fully mobilized and the posterior retrocardiac mediastinum completely exposed. The left and right pulmonary veins could be identified in the respective pulmonary hila but, when followed and carefully dissected, were noticed to be coursing posterior to the esophagus (Figure 1A). The esophagus was mobilized in its mid-portion, exposing the retroesophageal confluence; the reconstruction was then undertaken by vertically transecting the pulmonary venous confluence, yielding two pulmonary venous ostia measuring approximately 5-6 mm diameter. Each half of the divided confluence was incised on its anterior aspect and spatulated anastomoses were constructed. The left venous confluence was anastomosed directly onto the left atrial appendage with a 7-0 running polypropylene suture (modified Warden procedure); the right atrium was incised transversely across the interatrial septum and onto the posterior left atrial wall. The right venous confluence was anastomosed to the posterior transverse atriotomy (Figure 1B), and the atrial septal defect was closed with a patch of autologous pericardium . The postoperative course was uneventful, and the infant was discharged home on the tenth postoperative day. The child is alive and well with no residual pulmonary venous obstruction 3 years following the initial operation.
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The near totality of cases of TAPVC can be accounted for within the classification scheme proposed by Darling et al. in 1957.[1] According to this anatomic classification, only 5% of patients present with “unusual” morphology, including mixed (Type IV) or non-confluent veins. Several “unusual” patterns have been described[2,3] but, to our knowledge, no case of retroesophageal pulmonary venous confluence has thus far been reported. In mixed types without obstruction or with complex associated cardiac anomalies it might be beneficial to further delineate the anatomy with diagnostic imaging to aid in operative planning. In the case reported, the patient was correctly diagnosed with non-obstructed supracardiac TAPVC, although the confluence’s relationship with the esophagus could not be accurately delineated by preoperative transthoracic echocardiography.
ACCEPTED MANUSCRIPT
SC
RI PT
Intraoperatively, even transection of the inferior vena cava did not aid in elucidating the anatomy, and only antegrade dissection of the veins from the respective hila was able to disclose the site of obstruction behind the esophagus. Once the venous pattern was identified as joining behind the esophagus, two options were feasible to obtain unobstructed pulmonary venous return. The first would have been to perform a “sutureless repair” by incising the veins and the overlying parietal pericardium.[4] The dissection around the esophagus had by this decisional point been too extensive, leaving little pericardial integrity around the veins to allow for a hemostatic repair. We chose to instead individually anastomose the respective confluences to the left atrial appendage and to the posterior wall of the left atrium. Given the size of the vessels and the goal of minimizing the potential for late stenosis, we spatulated the anastomoses so as to maximize their linear length.
References
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1] DarlingRC, Rothney WB, Craig JM. Total anomalous pulmonary venous drainage into the right side of the heart. Lab Invest 1957;6:44. 2] Bharati S, Lev M. the pathology of congenital heart disease. A personal experience with more than 6,300 congenitally malformed hearts. Futura Publishing Company, Armonk, New York, 1996. Chapter 38: Total anomalous pulmonary venous drainage. 609-626.
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3] Cameron, D. Repair of unusual types of Total Anomalous Pulmonary Veins. Operative Techniques in Thoracic and Cardiovascular Surgery 2001;6(1):50.
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4] Yun TJ, Coles JG, Konstantinov I, Al-Radi OO, Wald RM, Guerra V, de Oliveira NC, Van Arsdell G, Williams WG, Smallhorn J, Caldarone CA. Conventional and sutureless techniques for management of the pulmonary veins: evolution of indications from postrepair pulmonary vein stenosis to primary pulmonary vein anomalies. J Thorac Cardiovasc Surg 2005;129(1):167.
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ACCEPTED MANUSCRIPT
Figure 1
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Figures
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A. Schematic, axial rendition of retroesophageal total anomalous pulmonary venous connection. (Esoph=esophagus, LA=left atrium, LAA=left atrial appendage, LLPV=left lower pulmonary vein, LUPV=left upper pulmonary vein, RA=right atrium, RLPV=right lower pulmonary vein, RUPV=right upper pulmonary vein, VV=vertical vein B. Following vertical transection of the retroesophageal confluence, the right veins are anastomosed to the posterior wall of the left atrium and the left veins to the left atrial appendage.
S0022-5223(14)00446-2
DOI:
10.1016/j.jtcvs.2014.04.022
Reference:
YMTC 8561
To appear in:
The Journal of Thoracic and Cardiovascular Surgery
Received Date: 8 April 2014 Accepted Date: 11 April 2014
Please cite this article as: Caldarone LA, Caldarone CA, Vricella LA, Retroesophageal Total Anomalous Pulmonary Venous Connection in a Newborn, The Journal of Thoracic and Cardiovascular Surgery (2014), doi: 10.1016/j.jtcvs.2014.04.022. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Retroesophageal Total Anomalous Pulmonary Venous Connection in a Newborn
RI PT
Lindsay A. Caldarone1, Christopher A. Caldarone2, Luca A. Vricella1
Division of Cardiac Surgery, Johns Hopkins University School of Medicine, Baltimore, USA1
AC C
EP
TE D
M AN U
SC
The Hospital for Sick Children, University of Toronto, Toronto, Canada2
Correspondence:
Luca A. Vricella, MD Director, Pediatric Cardiac Surgery and Heart Transplantation Johns Hopkins University, Baltimore, Maryland 1800 Orleans Street, Zayed 7107G Baltimore, MD 21287, USA Tel. (443) 287 - 1262 Fax (410) 955 - 3809 e-mail: [email protected]
WORD COUNT: 588
ACCEPTED MANUSCRIPT
Case Report
RI PT
A 2-month old, 4.3 Kg full-term infant with no prior cardiac diagnosis was found cyanotic and listless and emergently transported to our Institution, where a transthoracic echocardiogram revealed non-obstructed supracardiac total anomalous pulmonary venous connection (TAPVC) with a vertical vein coursing posterior to the left pulmonary artery and draining into the left brachiocephalic vein. The patient was brought to the operating room with the expectation that routine repair of supracardiac TAPVC would be performed.
Comment
EP
TE D
M AN U
SC
During surgery, however, the pulmonary venous confluence could not be identified through the posterior pericardium. Following cardioplegic and circulatory arrest at profound hypothermia, we attempted localization of the confluence and proceeded to even transect the inferior vena cava with the hope of finding the confluence once the heart could be fully mobilized and the posterior retrocardiac mediastinum completely exposed. The left and right pulmonary veins could be identified in the respective pulmonary hila but, when followed and carefully dissected, were noticed to be coursing posterior to the esophagus (Figure 1A). The esophagus was mobilized in its mid-portion, exposing the retroesophageal confluence; the reconstruction was then undertaken by vertically transecting the pulmonary venous confluence, yielding two pulmonary venous ostia measuring approximately 5-6 mm diameter. Each half of the divided confluence was incised on its anterior aspect and spatulated anastomoses were constructed. The left venous confluence was anastomosed directly onto the left atrial appendage with a 7-0 running polypropylene suture (modified Warden procedure); the right atrium was incised transversely across the interatrial septum and onto the posterior left atrial wall. The right venous confluence was anastomosed to the posterior transverse atriotomy (Figure 1B), and the atrial septal defect was closed with a patch of autologous pericardium . The postoperative course was uneventful, and the infant was discharged home on the tenth postoperative day. The child is alive and well with no residual pulmonary venous obstruction 3 years following the initial operation.
AC C
The near totality of cases of TAPVC can be accounted for within the classification scheme proposed by Darling et al. in 1957.[1] According to this anatomic classification, only 5% of patients present with “unusual” morphology, including mixed (Type IV) or non-confluent veins. Several “unusual” patterns have been described[2,3] but, to our knowledge, no case of retroesophageal pulmonary venous confluence has thus far been reported. In mixed types without obstruction or with complex associated cardiac anomalies it might be beneficial to further delineate the anatomy with diagnostic imaging to aid in operative planning. In the case reported, the patient was correctly diagnosed with non-obstructed supracardiac TAPVC, although the confluence’s relationship with the esophagus could not be accurately delineated by preoperative transthoracic echocardiography.
ACCEPTED MANUSCRIPT
SC
RI PT
Intraoperatively, even transection of the inferior vena cava did not aid in elucidating the anatomy, and only antegrade dissection of the veins from the respective hila was able to disclose the site of obstruction behind the esophagus. Once the venous pattern was identified as joining behind the esophagus, two options were feasible to obtain unobstructed pulmonary venous return. The first would have been to perform a “sutureless repair” by incising the veins and the overlying parietal pericardium.[4] The dissection around the esophagus had by this decisional point been too extensive, leaving little pericardial integrity around the veins to allow for a hemostatic repair. We chose to instead individually anastomose the respective confluences to the left atrial appendage and to the posterior wall of the left atrium. Given the size of the vessels and the goal of minimizing the potential for late stenosis, we spatulated the anastomoses so as to maximize their linear length.
References
M AN U
1] DarlingRC, Rothney WB, Craig JM. Total anomalous pulmonary venous drainage into the right side of the heart. Lab Invest 1957;6:44. 2] Bharati S, Lev M. the pathology of congenital heart disease. A personal experience with more than 6,300 congenitally malformed hearts. Futura Publishing Company, Armonk, New York, 1996. Chapter 38: Total anomalous pulmonary venous drainage. 609-626.
TE D
3] Cameron, D. Repair of unusual types of Total Anomalous Pulmonary Veins. Operative Techniques in Thoracic and Cardiovascular Surgery 2001;6(1):50.
AC C
EP
4] Yun TJ, Coles JG, Konstantinov I, Al-Radi OO, Wald RM, Guerra V, de Oliveira NC, Van Arsdell G, Williams WG, Smallhorn J, Caldarone CA. Conventional and sutureless techniques for management of the pulmonary veins: evolution of indications from postrepair pulmonary vein stenosis to primary pulmonary vein anomalies. J Thorac Cardiovasc Surg 2005;129(1):167.
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
Figure 1
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Figures
AC C
A. Schematic, axial rendition of retroesophageal total anomalous pulmonary venous connection. (Esoph=esophagus, LA=left atrium, LAA=left atrial appendage, LLPV=left lower pulmonary vein, LUPV=left upper pulmonary vein, RA=right atrium, RLPV=right lower pulmonary vein, RUPV=right upper pulmonary vein, VV=vertical vein B. Following vertical transection of the retroesophageal confluence, the right veins are anastomosed to the posterior wall of the left atrium and the left veins to the left atrial appendage.
