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Anatomic Resection to Manage Donor Partial Anomalous Pulmonary Venous Return During Lung Transplantation: A Case Report and Review
Anatomic Resection to Manage Donor Partial Anomalous Pulmonary Venous Return During Lung Transplantation: A Case Report and Review J.N. Melvan, S.D. Force, and M.S. Sancheti* Department of Cardiothoracic Surgery and Division of Cardiothoracic Surgery, Emory University School of Medicine, Atlanta, Georgia
ABSTRACT Introduction. Rare vascular malformations are discovered infrequently in donor lungs before implantation into recipients. However, the proper handling of such malformations can influence ischemic time, implantation strategies, and subsequent patient outcomes. Case Report. We report a simplified method for addressing the technical challenges of anomalous pulmonary venous return in a donor lung before implantation. Conclusions. We demonstrate that anatomic resection is a safe and efficient method for managing this rare congenital vascular malformation.
A
NOMALOUS PULMONARY VENOUS RETURN (APVR) is a rare congenital malformation of the pulmonary vasculature. Seldom are these aberrations evident clinically; rather, are most often discovered in donor lungs post mortem. Abnormal pulmonary vasculature, like APVR, in donor lung tissue is challenging for transplant surgeons. Previous cases describe time-consuming, complex reconstruction techniques of the donor vasculature before implantation. We describe anatomic resection as a safe, efficient surgical solution to manage APVR in donor lungs during transplantation.
on the donor lung before implantation into the recipient. The middle/lower lobes of the donor right lung were then implanted in a conventional manner. The patient did well postoperatively with excellent graft expansion and function. He was discharged home on postoperative day 10 on room air. Recent pulmonary function tests, forced vital capacity of 2.69 (52% of predicted), forced expiratory volume in 1 second of 1.75 (45% of predicted), and carbon monoxide diffusing capacity of 17.4 (59% of predicted) and CT imaging (Fig 2) demonstrate that, at 2 years postoperatively the graft is performing well and clinically the patient continues to oxygenate well on room air with minimal dyspnea.
CASE REPORT
DISCUSSION
A 55-year-old man with end-stage lung disease secondary to idiopathic pulmonary fibrosis underwent bilateral sequential lung transplantation. The donor was a 42-year-old man who died from complications of end-stage renal disease without a history of cardiopulmonary disease. All standard donor lung criteria were met before recovery of organs and the left donor lung with normal anatomy was implanted without complication. Unexpectedly during preparation of the right donor lung, a single, large separate pulmonary vein was identified draining the right upper lobe (Fig 1). This vein did not connect to the left atrium and instead drained into the superior vena cava. Because this vein was located anterior and superior to the pulmonary artery, we felt that an anastomosis between the aberrant vein and the left atrium would most likely compress the pulmonary artery. Therefore, we performed an anatomic right upper lobectomy
Partial APVR is a rare congenital anomaly in which venous drainage from aberrant vein(s) returns to the superior vena cava or right atrium instead of the left atrium. This pulmonary malformation is rarely seen in adults, with reported incidence of 0.5%e0.7% [1]. Because most occurrences are clinically insignificant, these occurrence rates are likely underestimated [2].
0041-1345/15 http://dx.doi.org/10.1016/j.transproceed.2015.02.010
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*Address correspondence to Manu Sancheti, MD, Assistant Professor of Surgery, Emory University School of Medicine, General Thoracic Surgery e Emory St. Joseph’s Hospital, 5665 Peachtree Dunwoody Rd NE, Suite 200, Atlanta, Georgia, 30342. E-mail: [email protected] ª 2015 by Elsevier Inc. All rights reserved. 360 Park Avenue South, New York, NY 10010-1710
Transplantation Proceedings, 47, 846e848 (2015)
DONOR ANOMALOUS PULMONARY VENOUS RETURN
847
Fig 1. Resected donor right upper lobe with isolated anomalous pulmonary vein.
APVR results from failure of the primordial cardiac and pulmonary systems to fuse during embryologic development, normally occurring by 40 days’ gestation. Subsequently, this pulmonary anomaly is often associated with additional cardiac defects and congenital syndromes, including Turner and Scimitar syndromes [3]. Aberrant vessels originate from the right lung in 92%e97% of cases [1]. APVR can be corrected surgically in symptomatic pediatric patients. The most common variant described in 80% of cases, and as seen in our patient, is flow from the right superior pulmonary vein into the superior vena cava [2]. If APVR persists into adulthood, it can manifest with symptoms of lefteright heart shunting, pulmonary artery hypertension, right ventricular volume overload, and resultant development of right ventricular failure [3].
Fig 2. Pretransplant (A, axial; B, coronal) and posttransplant (C, axial; D, coronal) CT of the chest.
Management of APVR during lung transplantation has been described previously using complex reconstruction techniques in which pericardial and iliac vein conduits [4] as well as the direct anastomosis of aberrant vessels to atrial appendages are used to overcome the abnormal vascular anatomy [5]. These techniques can be time consuming and add risks of obstruction, stenosis, and reoperation [3]. We present the first reported case of anatomic pulmonary resection to manage APVR during lung transplantation. Anatomic resection is commonly employed by thoracic surgeons and can circumvent potentially time-consuming, complex reconstruction techniques during transplantation. Although we do not advocate for superiority, we show that this simple technique, performed owing to anatomic limitations in our patient, was well-tolerated with adequate preservation of
848
lung parenchyma. Therefore, we believe that anatomic resection should be considered safe and an alternative way to handle donor lobar anomalies in patients undergoing bilateral lung transplantation.
REFERENCES [1] Gustafson RA, Warden HE, Murray GF, et al. Partial anomalous pulmonary venous connection to the right side of the heart. J Thorac Cardiovasc Surg 1989;98:861e8.
MELVAN, FORCE, AND SANCHETI [2] Alpert JS, Dexter L, Vieweg WV, et al. Anomalous pulmonary venous return with intact atrial septum: diagnosis and pathophysiology. Circulation 1977;56:870e5. [3] Sears EH, Aliotta JM, Klinger JR. Partial anomalous pulmonary venous return presenting with adult-onset pulmonary hypertension. Pulm Circ 2012;2:250e5. [4] Schmidt F, McGiffin DC, Zorn G, et al. Management of congenital abnormalities of the donor lung. Ann Thorac Surg 2001;72:935e7. [5] Khasati NH, MacHaal A, Thekkudan J, et al. An aberrant donor pulmonary vein during lung transplant: a surgical challenge. Ann Thorac Surg 2005;79:330e1.
ABSTRACT Introduction. Rare vascular malformations are discovered infrequently in donor lungs before implantation into recipients. However, the proper handling of such malformations can influence ischemic time, implantation strategies, and subsequent patient outcomes. Case Report. We report a simplified method for addressing the technical challenges of anomalous pulmonary venous return in a donor lung before implantation. Conclusions. We demonstrate that anatomic resection is a safe and efficient method for managing this rare congenital vascular malformation.
A
NOMALOUS PULMONARY VENOUS RETURN (APVR) is a rare congenital malformation of the pulmonary vasculature. Seldom are these aberrations evident clinically; rather, are most often discovered in donor lungs post mortem. Abnormal pulmonary vasculature, like APVR, in donor lung tissue is challenging for transplant surgeons. Previous cases describe time-consuming, complex reconstruction techniques of the donor vasculature before implantation. We describe anatomic resection as a safe, efficient surgical solution to manage APVR in donor lungs during transplantation.
on the donor lung before implantation into the recipient. The middle/lower lobes of the donor right lung were then implanted in a conventional manner. The patient did well postoperatively with excellent graft expansion and function. He was discharged home on postoperative day 10 on room air. Recent pulmonary function tests, forced vital capacity of 2.69 (52% of predicted), forced expiratory volume in 1 second of 1.75 (45% of predicted), and carbon monoxide diffusing capacity of 17.4 (59% of predicted) and CT imaging (Fig 2) demonstrate that, at 2 years postoperatively the graft is performing well and clinically the patient continues to oxygenate well on room air with minimal dyspnea.
CASE REPORT
DISCUSSION
A 55-year-old man with end-stage lung disease secondary to idiopathic pulmonary fibrosis underwent bilateral sequential lung transplantation. The donor was a 42-year-old man who died from complications of end-stage renal disease without a history of cardiopulmonary disease. All standard donor lung criteria were met before recovery of organs and the left donor lung with normal anatomy was implanted without complication. Unexpectedly during preparation of the right donor lung, a single, large separate pulmonary vein was identified draining the right upper lobe (Fig 1). This vein did not connect to the left atrium and instead drained into the superior vena cava. Because this vein was located anterior and superior to the pulmonary artery, we felt that an anastomosis between the aberrant vein and the left atrium would most likely compress the pulmonary artery. Therefore, we performed an anatomic right upper lobectomy
Partial APVR is a rare congenital anomaly in which venous drainage from aberrant vein(s) returns to the superior vena cava or right atrium instead of the left atrium. This pulmonary malformation is rarely seen in adults, with reported incidence of 0.5%e0.7% [1]. Because most occurrences are clinically insignificant, these occurrence rates are likely underestimated [2].
0041-1345/15 http://dx.doi.org/10.1016/j.transproceed.2015.02.010
846
*Address correspondence to Manu Sancheti, MD, Assistant Professor of Surgery, Emory University School of Medicine, General Thoracic Surgery e Emory St. Joseph’s Hospital, 5665 Peachtree Dunwoody Rd NE, Suite 200, Atlanta, Georgia, 30342. E-mail: [email protected] ª 2015 by Elsevier Inc. All rights reserved. 360 Park Avenue South, New York, NY 10010-1710
Transplantation Proceedings, 47, 846e848 (2015)
DONOR ANOMALOUS PULMONARY VENOUS RETURN
847
Fig 1. Resected donor right upper lobe with isolated anomalous pulmonary vein.
APVR results from failure of the primordial cardiac and pulmonary systems to fuse during embryologic development, normally occurring by 40 days’ gestation. Subsequently, this pulmonary anomaly is often associated with additional cardiac defects and congenital syndromes, including Turner and Scimitar syndromes [3]. Aberrant vessels originate from the right lung in 92%e97% of cases [1]. APVR can be corrected surgically in symptomatic pediatric patients. The most common variant described in 80% of cases, and as seen in our patient, is flow from the right superior pulmonary vein into the superior vena cava [2]. If APVR persists into adulthood, it can manifest with symptoms of lefteright heart shunting, pulmonary artery hypertension, right ventricular volume overload, and resultant development of right ventricular failure [3].
Fig 2. Pretransplant (A, axial; B, coronal) and posttransplant (C, axial; D, coronal) CT of the chest.
Management of APVR during lung transplantation has been described previously using complex reconstruction techniques in which pericardial and iliac vein conduits [4] as well as the direct anastomosis of aberrant vessels to atrial appendages are used to overcome the abnormal vascular anatomy [5]. These techniques can be time consuming and add risks of obstruction, stenosis, and reoperation [3]. We present the first reported case of anatomic pulmonary resection to manage APVR during lung transplantation. Anatomic resection is commonly employed by thoracic surgeons and can circumvent potentially time-consuming, complex reconstruction techniques during transplantation. Although we do not advocate for superiority, we show that this simple technique, performed owing to anatomic limitations in our patient, was well-tolerated with adequate preservation of
848
lung parenchyma. Therefore, we believe that anatomic resection should be considered safe and an alternative way to handle donor lobar anomalies in patients undergoing bilateral lung transplantation.
REFERENCES [1] Gustafson RA, Warden HE, Murray GF, et al. Partial anomalous pulmonary venous connection to the right side of the heart. J Thorac Cardiovasc Surg 1989;98:861e8.
MELVAN, FORCE, AND SANCHETI [2] Alpert JS, Dexter L, Vieweg WV, et al. Anomalous pulmonary venous return with intact atrial septum: diagnosis and pathophysiology. Circulation 1977;56:870e5. [3] Sears EH, Aliotta JM, Klinger JR. Partial anomalous pulmonary venous return presenting with adult-onset pulmonary hypertension. Pulm Circ 2012;2:250e5. [4] Schmidt F, McGiffin DC, Zorn G, et al. Management of congenital abnormalities of the donor lung. Ann Thorac Surg 2001;72:935e7. [5] Khasati NH, MacHaal A, Thekkudan J, et al. An aberrant donor pulmonary vein during lung transplant: a surgical challenge. Ann Thorac Surg 2005;79:330e1.