- Email: [email protected]
Isolated left ventricular ischemia after the Norwood procedure
Ann Thorac Surg 2002;73:657–9
CASE REPORT DeROSE ET AL LEFT VENTRICULAR ISCHEMIA AFTER NORWOOD PROCEDURE
repair has been performed. Both pericardium and right atrium have been used successfully as intraatrial baffles. A few points concerning operative technique deserve mention. In construction of the systemic venous baffle, the atrial septal defect must be large enough to allow for unimpeded flow into the anatomic left atrium. In the supracardiac type of total anomalous pulmonary venous return, if adequate tissue is present, the left atrial appendage can be anastomosed directly to the pulmonary venous confluence. If not, or if the confluence does not appear large enough to carry the venous return from both lungs, as in this report, pericardial enlargement can be used. The authors are grateful to Terry Wyffels for the illustrations.
References 1. Yamagishi M, Nakamura Y, Kanazawa T, Kawada N. Double switch operation for corrected transposition with total anomalous pulmonary venous return. J Thorac Cardiovasc Surg 1997;114:848–50. 2. Ueda Y, Miki S, Okita Y, et al. Transposition of the great arteries associated with total anomalous pulmonary venous return. Ann Thorac Surg 1994;57:470–2. 3. Sapsford RN, Aberdeen E, Watson DA, Crew AD. Transposed great arteries combined with totally anomalous pulmonary veins: a report of a successful correction. J Thorac Cardiovasc Surg 1972;63:360– 6. 4. Amodeo A, Corno A, Marino B, Carta MG, Marcelletti C. Combined repair of transposed great arteries and total anomalous pulmonary venous connection. Ann Thorac Surg 1990; 50:820–1.
Isolated Left Ventricular Ischemia After the Norwood Procedure Joseph J. DeRose, Jr, MD, Rozelle Corda, NP, M. Renate Dische, MD, PhD, Jennifer Eleazar, MD, and Ralph S. Mosca, MD Division of Pediatric Cardiac Surgery and Department of Pathology, Columbia University College of Physicians and Surgeons, New York, New York
Aortic atresia is the most severe variant of hypoplastic left heart syndrome (HLHS), and has been associated with significant mortality after stage I palliation. Coronary artery abnormalities are more prominent in this group of patients, especially in the presence of a patent mitral valve. Herein, we describe a case of isolated left ventricular ischemia after the Norwood procedure in a neonate with hypoplastic left heart syndrome, left ventricular hypertrophy, mitral stenosis, aortic atresia, and anomalous left coronary artery. (Ann Thorac Surg 2002;73:657–9) © 2002 by The Society of Thoracic Surgeons Accepted for publication June 22, 2001. Address reprint requests to Dr Mosca, Pediatric Cardiac Surgery, Columbia-Presbyterian Medical Center, 3959 Broadway, BHN 274, New York, NY 10032; e-mail: [email protected].
© 2002 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
657
D
espite improvements in surgical technique and postoperative care, in-hospital mortality after stage I palliation for hypoplastic left heart syndrome (HLHS) remains high in most large series (15% to 25%) [1, 2]. Aortic atresia has been identified as a risk factor by some surgeons for decreased early and late survival after the Norwood procedure [3]. Coronary insufficiency can result from technical challenges associated with the anastomosis of the small ascending aorta, but intrinsic coronary abnormalities can further compromise myocardial perfusion [4 –7]. We describe a case of intractable left ventricular ischemia after the Norwood procedure in a patient with profound left ventricular hypertrophy and left coronary artery abnormalities. A 2.4 kg male infant born at 30 weeks gestation presented with cyanosis in the newborn nursery of a referring hospital. Transthoracic echocardiogram established the diagnosis of HLHS. The infant was begun on prostaglandin E and transferred to our institution for definitive treatment. Upon arrival, continued respiratory distress led to endotracheal intubation. A repeat echocardiogram revealed HLHS with marked left ventricular hypertrophy, mitral stenosis, aortic atresia, and a mildly restrictive atrial septal defect (Fig 1). The infant was taken to the operating room on day of life 8 without evidence of end-organ hypoperfusion. Cardiopulmonary was initiated through right atrial and pulmonary artery cannulation. The branch pulmonary arteries were snared and the infant was cooled to 18°C. The descending aorta was cross-clamped and cardioplegia was administered (15 mL/kg) with prompt arrest and cooling of the heart. The Norwood reconstruction was then performed under circulatory arrest as previously described. A 3.0-mm shunt was fashioned from the innominate artery to the pulmonary bifurcation during rewarming. After complete rewarming and an attempt to wean from cardiopulmonary bypass, it became evident that despite excellent right ventricular function and perfusion, the left ventricle was markedly ischemic. Although the left coronary artery did not appear compromised, the patient was again cooled to 20°C and the circulation arrested. The proximal anastomosis was taken down and the coronary arteries inspected from the inside. There was no evidence of kinking or intraluminal obstruction. The patient was rewarmed but isolated left ventricular hypoperfusion persisted. Despite good right ventricular perfusion and function, the patient could not be weaned from cardiopulmonary bypass with maximal inotropic support and died in the operating room. An autopsy limited to the heart and thoracic great vessels revealed severe left ventricular hypertrophy with aortic atresia, mitral valve stenosis, and mild focal endocardial sclerosis. The thick ventricular septum bulged markedly into the right ventricle encroaching on its lumen. The increased left ventricular wall thickness appeared secondary to an increase in myocardial cell number without evidence of myofiber enlargement. The coronary anastomosis was widely patent, but the coronary arteries feeding the left ventricle were quite abnormal and atretic. The 0003-4975/02/$22.00 PII S0003-4975(01)03112-5
658
CASE REPORT DeROSE ET AL LEFT VENTRICULAR ISCHEMIA AFTER NORWOOD PROCEDURE
Fig 1. Echocardiogram demonstrating small, hypoplastic left ventricular cavity with marked left ventricular muscular hypertrophy. (Asc Ao ⫽ ascending aorta; LA ⫽ left atrium; LV ⫽ left ventricle; RV ⫽ right ventricle.)
left coronary artery branched abnormally resulting in a significant steal of blood from the left ventricle. It gave rise to a small anterior descending artery, the circumflex artery, and an aberrant branch that coursed below the right pulmonary artery and behind the right atrium toward the right pulmonary hilum (Fig 2). The intramyocardial vessels demonstrated significant tortuosity and ventriculocoronary communications were not identified.
Ann Thorac Surg 2002;73:657–9
HLHS have little effect on both right ventricular perfusion and histology. However, the severe left ventricular and septal hypertrophy in this infant, coupled with ischemic dysfunction may have resulted in the catastrophic consequences of septal dyscoordination and right ventricular obstruction. In autopsy report 122 patients who died after the Norwood procedure, Bartram and associates [10] found impairment in coronary perfusion to be the cause of death in 27% of the cases. However, the cause of stenosis in the vast majority of these patients (31 of 33) was secondary to either intraluminal stenosis at the anastomosis or external kinking from the homograft. Only 1 patient exhibited coronary arterial hypoplasia. These findings support the fact that ischemic episodes after the Norwood operation are most frequently the result of technical errors, and a rigorous evaluation of the proximal coronary anastomosis including reinstitution of circulatory arrest is nearly always warranted. Nonetheless, HLHS characterized by aortic atresia/ mitral stenosis and profound left ventricular hypertrophy may represent a subset of patients who should undergo detailed angiography of the coronary circulation before embarking on Norwood palliation. If coronary abnormalities in such patients are found, serious consideration should be given to transplantation as an alternative therapeutic option.
Comment Coronary abnormalities have previously been described in patients with HLHS [4 –7]. Analogous to the patent inflow and obstructed outflow of pulmonary atresia with intact ventricular septum, HLHS characterized by mitral stenosis and aortic atresia carries the highest incidence of coronary abnormalities [5, 7]. Ventriculocoronary communications of the luminal subtype are the most common and are observed in approximately 10% to 20% of such patients [7]. These direct arterioluminal connections have intimal abnormalities ranging from mild thickening to severe stenosis or obliteration of the lumen. Epicardial coronary abnormalities are rare and include aberrant origin from the pulmonary artery [9], diffuse hypoplasia [6, 9], and tortuosity [5–7]. In the present case, two causes of potential coronary hypoperfusion exist: (1) left coronary hypoplasia in both the anterior descending and circumflex distribution, and (2) an abnormal coronary artery branch stealing blood flow from the myocardial circulation. Marked left ventricular wall thickness undoubtedly placed increased oxygen demands on the hypoplastic left coronary circulation. It is presumed that upon the institution of cardiopulmonary bypass a decrease in coronary perfusion pressure together with an increase in coronary steal from the aberrant coronary branch resulted in critical left ventricular ischemia. Baffa and colleagues [7] have documented that left ventricular coronary abnormalities in
Fig 2. Postmortem schematic drawing demonstrating aberrant coronary artery coursing posteriorly from the circumflex coronary artery to the right pulmonary hilum. (LA ⫽ left atrium; LAD ⫽ left anterior descending coronary artery; LV ⫽ left ventricle; OD ⫽ outer dimension; RA ⫽ right atrium; RCA ⫽ right coronary artery; RV ⫽ right ventricle.)
Ann Thorac Surg 2002;73:659 – 61 21
CASE REPORT TAKAYAMA ET AL AORTOPULMONARY WINDOW DUE TO BALLOON ANGIOPLASTY
References 1. Mosca RS, Bove EL, Crowley DC, Sandhu SK, Schork MA, Kulik TJ. Hemodynamic characteristics of neonates following first stage palliation for hypoplastic left heart syndrome. Circulation 1995;92(Suppl 2):267–71. 2. Kern JH, Hayes CJ, Michler RE, Gersony WM, Quaegebeur JM. Survival and risk analysis for the Norwood procedure for hypoplastic left heart syndrome. Am J Cardiol 1997;80:170– 4. 3. Jonas RA, Hanson DD, Cook N, Wessel D. Anatomic subtype and survival after reconstructive operation for hypoplastic left heart syndrome. J Thoracic Cardiovasc Surg 1994;107:1121– 8. 4. Beckman CB, Moller JH, Edwards JE. Alternate pathways to pulmonary venous flow in left-sided obstructive anomalies. Circulation 1975;52:509–16. 5. O’Connor WN, Cash JB, Cottrill CM, Johnson GL, Noonan JA. Ventriculo-coronary connections in hypoplastic left hearts: an autopsy microscopic study. Circulation 1982;66:1078– 86. 6. Sauer U, Gittenberger-de Groot AC, Geishauser M, Babic R, Buhlmeyer K. Coronary arteries in the hypoplastic left heart syndrome: Histopathologic and histometrical studies and implications for surgery. Circulation 1989;80(Suppl 1):I168–76. 7. Baffa JM, Chen SL, Guttenberg ME, Norwood WI, Weinberg PM. Coronary artery abnormalities and right ventricular histology in hypoplastic left heart syndrome. J Am Coll Cardiol 1992;20:350– 8. 8. Freedom RM, Culham JAC, Moes CAF, Harrington DP. Selective aortic root angiography in the hypoplastic left heart syndrome. Eur J Cardiol 1976;4:25–9. 9. Sarris GE, Drummond-Webb JJ, Ebdeid MR, Latson LA, Mee RB. Anomalous origin of left coronary artery from the right pulmonary artery in hypoplastic left heart syndrome. Ann Thorac Surg 1997;64:836– 8. 10. Bartram U, Grunenfelder J, van Praagh R. Causes of death after the modified Norwood procedure: a study of 122 postmortem cases. Ann Thorac Surg 1997;64:1795– 802.
Aortopulmonary Window Due to Balloon Angioplasty After Arterial Switch Operation Hiroo Takayama, MD, Akihiko Sekiguchi, MD, Masahide Chikada, MD, Mio Noma, MD, and Ryoichi Ishida, MD Division of Cardiovascular Surgery, National Children’s Hospital, Tokyo, Japan
We report 2 cases of aortopulmonary window that developed after balloon angioplasty for pulmonary artery stenosis. Both patients had undergone arterial switch operations for complete transposition of the great arteries before the angioplasty. These aortopulmonary windows were repaired through elective operations. The clinical features, diagnosis, management, and proposed mechanisms of this complication are described. (Ann Thorac Surg 2002;73:659 – 61) © 2002 by The Society of Thoracic Surgeons
A
ortopulmonary (AP) window as a complication of pulmonary artery (PA) balloon angioplasty has rarely been reported [1]. We report 2 cases of traumatic
Accepted for publication June 25, 2001. Address reprint requests to Dr Takayama, Division of Cardiovascular Surgery, National Children’s Hospital, 3-35-31 Taishido Setagaya-ku, Tokyo, 154-8509, Japan; e-mail: [email protected].
© 2002 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
659
AP window, both of which developed in patients after an arterial switch operation (ASO).
Case Reports Patient 1 A female newborn with d-transposition of the great arteries with intact ventricular septum underwent an ASO on day 15 of life. Follow-up cardiac catheterization demonstrated bilateral PA stenosis, which was 4.4 mm at the right and 6.0 mm at the left in diameter. Fifteen months after the operation, serial dilations were successfully performed under the pressure of 10 atmospheres using 12-mm Ultrathin diamond balloon (Boston Scientific Corporation, Natick, MA). Echocardiography after the procedure demonstrated AP communication at PA bifurcation, although pulmonary angiography immediately after the dilation showed no such findings (Fig 1). Because the patient was hemodynamically stable, evaluation catheterization was performed 6 months after the intervention. Oxymetric data revealed a step-up in oxygen saturation in the right PA, and pulmonic-to-systemic ratio (Qp/Qs) was calculated to be 1.56. The AP window was confirmed by ascending aortography. The patient underwent surgery for this defect 21 months after the balloon dilation. A defect that was oval in shape and 5 mm at greatest diameter was examined through a pulmonary arteriotomy (Fig 2). The surgical procedure included direct closure of this defect and patch angioplasty of PA bifurcation.
Patient 2 A male newborn diagnosed with d-transposition of the great arteries with intact ventricular septum underwent left modified Blalock-Taussig shunt and PA banding at 3 months, as well as ASO at 9 months. The patient developed bilateral PA stenosis, and underwent dilation with balloon angioplasty 3 years after ASO. Follow-up cardiac catheterization demonstrated restenosis of right PA, which was 3.7 mm in diameter. Balloon angioplasty was attempted with 12-mm Blue-Max balloon catheter (Boston Scientific Corporation, Natick, MA) 3.5 years after ASO. At the time of this procedure, the patient collapsed hemodynamically and was resuscitated. Chest roentgenography revealed abnormal shadow at the hilar portion of right lung. The patient was observed closely under the suspicion of a PA rupture, and recovered with conservative treatment. Cardiac catheterization 5 months after this intervention demonstrated a communication between the aorta and right PA. Oxymetric data revealed a step-up in oxygen saturation in right PA. The Qp/Qs was calculated to be 1.78. Surgery was attempted 21 months after the balloon angioplasty. A defect 5 mm in diameter was found between the two arteries. The edge of this defect was so friable that the defect needed to be closed with a patch. The PA bifurcation was enlarged using an autologous pericardial patch. Follow-up cardiac catheterization demonstrated a residual communication around the closure site. 0003-4975/02/$22.00 PII S0003-4975(01)03113-7
CASE REPORT DeROSE ET AL LEFT VENTRICULAR ISCHEMIA AFTER NORWOOD PROCEDURE
repair has been performed. Both pericardium and right atrium have been used successfully as intraatrial baffles. A few points concerning operative technique deserve mention. In construction of the systemic venous baffle, the atrial septal defect must be large enough to allow for unimpeded flow into the anatomic left atrium. In the supracardiac type of total anomalous pulmonary venous return, if adequate tissue is present, the left atrial appendage can be anastomosed directly to the pulmonary venous confluence. If not, or if the confluence does not appear large enough to carry the venous return from both lungs, as in this report, pericardial enlargement can be used. The authors are grateful to Terry Wyffels for the illustrations.
References 1. Yamagishi M, Nakamura Y, Kanazawa T, Kawada N. Double switch operation for corrected transposition with total anomalous pulmonary venous return. J Thorac Cardiovasc Surg 1997;114:848–50. 2. Ueda Y, Miki S, Okita Y, et al. Transposition of the great arteries associated with total anomalous pulmonary venous return. Ann Thorac Surg 1994;57:470–2. 3. Sapsford RN, Aberdeen E, Watson DA, Crew AD. Transposed great arteries combined with totally anomalous pulmonary veins: a report of a successful correction. J Thorac Cardiovasc Surg 1972;63:360– 6. 4. Amodeo A, Corno A, Marino B, Carta MG, Marcelletti C. Combined repair of transposed great arteries and total anomalous pulmonary venous connection. Ann Thorac Surg 1990; 50:820–1.
Isolated Left Ventricular Ischemia After the Norwood Procedure Joseph J. DeRose, Jr, MD, Rozelle Corda, NP, M. Renate Dische, MD, PhD, Jennifer Eleazar, MD, and Ralph S. Mosca, MD Division of Pediatric Cardiac Surgery and Department of Pathology, Columbia University College of Physicians and Surgeons, New York, New York
Aortic atresia is the most severe variant of hypoplastic left heart syndrome (HLHS), and has been associated with significant mortality after stage I palliation. Coronary artery abnormalities are more prominent in this group of patients, especially in the presence of a patent mitral valve. Herein, we describe a case of isolated left ventricular ischemia after the Norwood procedure in a neonate with hypoplastic left heart syndrome, left ventricular hypertrophy, mitral stenosis, aortic atresia, and anomalous left coronary artery. (Ann Thorac Surg 2002;73:657–9) © 2002 by The Society of Thoracic Surgeons Accepted for publication June 22, 2001. Address reprint requests to Dr Mosca, Pediatric Cardiac Surgery, Columbia-Presbyterian Medical Center, 3959 Broadway, BHN 274, New York, NY 10032; e-mail: [email protected].
© 2002 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
657
D
espite improvements in surgical technique and postoperative care, in-hospital mortality after stage I palliation for hypoplastic left heart syndrome (HLHS) remains high in most large series (15% to 25%) [1, 2]. Aortic atresia has been identified as a risk factor by some surgeons for decreased early and late survival after the Norwood procedure [3]. Coronary insufficiency can result from technical challenges associated with the anastomosis of the small ascending aorta, but intrinsic coronary abnormalities can further compromise myocardial perfusion [4 –7]. We describe a case of intractable left ventricular ischemia after the Norwood procedure in a patient with profound left ventricular hypertrophy and left coronary artery abnormalities. A 2.4 kg male infant born at 30 weeks gestation presented with cyanosis in the newborn nursery of a referring hospital. Transthoracic echocardiogram established the diagnosis of HLHS. The infant was begun on prostaglandin E and transferred to our institution for definitive treatment. Upon arrival, continued respiratory distress led to endotracheal intubation. A repeat echocardiogram revealed HLHS with marked left ventricular hypertrophy, mitral stenosis, aortic atresia, and a mildly restrictive atrial septal defect (Fig 1). The infant was taken to the operating room on day of life 8 without evidence of end-organ hypoperfusion. Cardiopulmonary was initiated through right atrial and pulmonary artery cannulation. The branch pulmonary arteries were snared and the infant was cooled to 18°C. The descending aorta was cross-clamped and cardioplegia was administered (15 mL/kg) with prompt arrest and cooling of the heart. The Norwood reconstruction was then performed under circulatory arrest as previously described. A 3.0-mm shunt was fashioned from the innominate artery to the pulmonary bifurcation during rewarming. After complete rewarming and an attempt to wean from cardiopulmonary bypass, it became evident that despite excellent right ventricular function and perfusion, the left ventricle was markedly ischemic. Although the left coronary artery did not appear compromised, the patient was again cooled to 20°C and the circulation arrested. The proximal anastomosis was taken down and the coronary arteries inspected from the inside. There was no evidence of kinking or intraluminal obstruction. The patient was rewarmed but isolated left ventricular hypoperfusion persisted. Despite good right ventricular perfusion and function, the patient could not be weaned from cardiopulmonary bypass with maximal inotropic support and died in the operating room. An autopsy limited to the heart and thoracic great vessels revealed severe left ventricular hypertrophy with aortic atresia, mitral valve stenosis, and mild focal endocardial sclerosis. The thick ventricular septum bulged markedly into the right ventricle encroaching on its lumen. The increased left ventricular wall thickness appeared secondary to an increase in myocardial cell number without evidence of myofiber enlargement. The coronary anastomosis was widely patent, but the coronary arteries feeding the left ventricle were quite abnormal and atretic. The 0003-4975/02/$22.00 PII S0003-4975(01)03112-5
658
CASE REPORT DeROSE ET AL LEFT VENTRICULAR ISCHEMIA AFTER NORWOOD PROCEDURE
Fig 1. Echocardiogram demonstrating small, hypoplastic left ventricular cavity with marked left ventricular muscular hypertrophy. (Asc Ao ⫽ ascending aorta; LA ⫽ left atrium; LV ⫽ left ventricle; RV ⫽ right ventricle.)
left coronary artery branched abnormally resulting in a significant steal of blood from the left ventricle. It gave rise to a small anterior descending artery, the circumflex artery, and an aberrant branch that coursed below the right pulmonary artery and behind the right atrium toward the right pulmonary hilum (Fig 2). The intramyocardial vessels demonstrated significant tortuosity and ventriculocoronary communications were not identified.
Ann Thorac Surg 2002;73:657–9
HLHS have little effect on both right ventricular perfusion and histology. However, the severe left ventricular and septal hypertrophy in this infant, coupled with ischemic dysfunction may have resulted in the catastrophic consequences of septal dyscoordination and right ventricular obstruction. In autopsy report 122 patients who died after the Norwood procedure, Bartram and associates [10] found impairment in coronary perfusion to be the cause of death in 27% of the cases. However, the cause of stenosis in the vast majority of these patients (31 of 33) was secondary to either intraluminal stenosis at the anastomosis or external kinking from the homograft. Only 1 patient exhibited coronary arterial hypoplasia. These findings support the fact that ischemic episodes after the Norwood operation are most frequently the result of technical errors, and a rigorous evaluation of the proximal coronary anastomosis including reinstitution of circulatory arrest is nearly always warranted. Nonetheless, HLHS characterized by aortic atresia/ mitral stenosis and profound left ventricular hypertrophy may represent a subset of patients who should undergo detailed angiography of the coronary circulation before embarking on Norwood palliation. If coronary abnormalities in such patients are found, serious consideration should be given to transplantation as an alternative therapeutic option.
Comment Coronary abnormalities have previously been described in patients with HLHS [4 –7]. Analogous to the patent inflow and obstructed outflow of pulmonary atresia with intact ventricular septum, HLHS characterized by mitral stenosis and aortic atresia carries the highest incidence of coronary abnormalities [5, 7]. Ventriculocoronary communications of the luminal subtype are the most common and are observed in approximately 10% to 20% of such patients [7]. These direct arterioluminal connections have intimal abnormalities ranging from mild thickening to severe stenosis or obliteration of the lumen. Epicardial coronary abnormalities are rare and include aberrant origin from the pulmonary artery [9], diffuse hypoplasia [6, 9], and tortuosity [5–7]. In the present case, two causes of potential coronary hypoperfusion exist: (1) left coronary hypoplasia in both the anterior descending and circumflex distribution, and (2) an abnormal coronary artery branch stealing blood flow from the myocardial circulation. Marked left ventricular wall thickness undoubtedly placed increased oxygen demands on the hypoplastic left coronary circulation. It is presumed that upon the institution of cardiopulmonary bypass a decrease in coronary perfusion pressure together with an increase in coronary steal from the aberrant coronary branch resulted in critical left ventricular ischemia. Baffa and colleagues [7] have documented that left ventricular coronary abnormalities in
Fig 2. Postmortem schematic drawing demonstrating aberrant coronary artery coursing posteriorly from the circumflex coronary artery to the right pulmonary hilum. (LA ⫽ left atrium; LAD ⫽ left anterior descending coronary artery; LV ⫽ left ventricle; OD ⫽ outer dimension; RA ⫽ right atrium; RCA ⫽ right coronary artery; RV ⫽ right ventricle.)
Ann Thorac Surg 2002;73:659 – 61 21
CASE REPORT TAKAYAMA ET AL AORTOPULMONARY WINDOW DUE TO BALLOON ANGIOPLASTY
References 1. Mosca RS, Bove EL, Crowley DC, Sandhu SK, Schork MA, Kulik TJ. Hemodynamic characteristics of neonates following first stage palliation for hypoplastic left heart syndrome. Circulation 1995;92(Suppl 2):267–71. 2. Kern JH, Hayes CJ, Michler RE, Gersony WM, Quaegebeur JM. Survival and risk analysis for the Norwood procedure for hypoplastic left heart syndrome. Am J Cardiol 1997;80:170– 4. 3. Jonas RA, Hanson DD, Cook N, Wessel D. Anatomic subtype and survival after reconstructive operation for hypoplastic left heart syndrome. J Thoracic Cardiovasc Surg 1994;107:1121– 8. 4. Beckman CB, Moller JH, Edwards JE. Alternate pathways to pulmonary venous flow in left-sided obstructive anomalies. Circulation 1975;52:509–16. 5. O’Connor WN, Cash JB, Cottrill CM, Johnson GL, Noonan JA. Ventriculo-coronary connections in hypoplastic left hearts: an autopsy microscopic study. Circulation 1982;66:1078– 86. 6. Sauer U, Gittenberger-de Groot AC, Geishauser M, Babic R, Buhlmeyer K. Coronary arteries in the hypoplastic left heart syndrome: Histopathologic and histometrical studies and implications for surgery. Circulation 1989;80(Suppl 1):I168–76. 7. Baffa JM, Chen SL, Guttenberg ME, Norwood WI, Weinberg PM. Coronary artery abnormalities and right ventricular histology in hypoplastic left heart syndrome. J Am Coll Cardiol 1992;20:350– 8. 8. Freedom RM, Culham JAC, Moes CAF, Harrington DP. Selective aortic root angiography in the hypoplastic left heart syndrome. Eur J Cardiol 1976;4:25–9. 9. Sarris GE, Drummond-Webb JJ, Ebdeid MR, Latson LA, Mee RB. Anomalous origin of left coronary artery from the right pulmonary artery in hypoplastic left heart syndrome. Ann Thorac Surg 1997;64:836– 8. 10. Bartram U, Grunenfelder J, van Praagh R. Causes of death after the modified Norwood procedure: a study of 122 postmortem cases. Ann Thorac Surg 1997;64:1795– 802.
Aortopulmonary Window Due to Balloon Angioplasty After Arterial Switch Operation Hiroo Takayama, MD, Akihiko Sekiguchi, MD, Masahide Chikada, MD, Mio Noma, MD, and Ryoichi Ishida, MD Division of Cardiovascular Surgery, National Children’s Hospital, Tokyo, Japan
We report 2 cases of aortopulmonary window that developed after balloon angioplasty for pulmonary artery stenosis. Both patients had undergone arterial switch operations for complete transposition of the great arteries before the angioplasty. These aortopulmonary windows were repaired through elective operations. The clinical features, diagnosis, management, and proposed mechanisms of this complication are described. (Ann Thorac Surg 2002;73:659 – 61) © 2002 by The Society of Thoracic Surgeons
A
ortopulmonary (AP) window as a complication of pulmonary artery (PA) balloon angioplasty has rarely been reported [1]. We report 2 cases of traumatic
Accepted for publication June 25, 2001. Address reprint requests to Dr Takayama, Division of Cardiovascular Surgery, National Children’s Hospital, 3-35-31 Taishido Setagaya-ku, Tokyo, 154-8509, Japan; e-mail: [email protected].
© 2002 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
659
AP window, both of which developed in patients after an arterial switch operation (ASO).
Case Reports Patient 1 A female newborn with d-transposition of the great arteries with intact ventricular septum underwent an ASO on day 15 of life. Follow-up cardiac catheterization demonstrated bilateral PA stenosis, which was 4.4 mm at the right and 6.0 mm at the left in diameter. Fifteen months after the operation, serial dilations were successfully performed under the pressure of 10 atmospheres using 12-mm Ultrathin diamond balloon (Boston Scientific Corporation, Natick, MA). Echocardiography after the procedure demonstrated AP communication at PA bifurcation, although pulmonary angiography immediately after the dilation showed no such findings (Fig 1). Because the patient was hemodynamically stable, evaluation catheterization was performed 6 months after the intervention. Oxymetric data revealed a step-up in oxygen saturation in the right PA, and pulmonic-to-systemic ratio (Qp/Qs) was calculated to be 1.56. The AP window was confirmed by ascending aortography. The patient underwent surgery for this defect 21 months after the balloon dilation. A defect that was oval in shape and 5 mm at greatest diameter was examined through a pulmonary arteriotomy (Fig 2). The surgical procedure included direct closure of this defect and patch angioplasty of PA bifurcation.
Patient 2 A male newborn diagnosed with d-transposition of the great arteries with intact ventricular septum underwent left modified Blalock-Taussig shunt and PA banding at 3 months, as well as ASO at 9 months. The patient developed bilateral PA stenosis, and underwent dilation with balloon angioplasty 3 years after ASO. Follow-up cardiac catheterization demonstrated restenosis of right PA, which was 3.7 mm in diameter. Balloon angioplasty was attempted with 12-mm Blue-Max balloon catheter (Boston Scientific Corporation, Natick, MA) 3.5 years after ASO. At the time of this procedure, the patient collapsed hemodynamically and was resuscitated. Chest roentgenography revealed abnormal shadow at the hilar portion of right lung. The patient was observed closely under the suspicion of a PA rupture, and recovered with conservative treatment. Cardiac catheterization 5 months after this intervention demonstrated a communication between the aorta and right PA. Oxymetric data revealed a step-up in oxygen saturation in right PA. The Qp/Qs was calculated to be 1.78. Surgery was attempted 21 months after the balloon angioplasty. A defect 5 mm in diameter was found between the two arteries. The edge of this defect was so friable that the defect needed to be closed with a patch. The PA bifurcation was enlarged using an autologous pericardial patch. Follow-up cardiac catheterization demonstrated a residual communication around the closure site. 0003-4975/02/$22.00 PII S0003-4975(01)03113-7