- Email: [email protected]
Aortopulmonary Window and Anomalous Origin of the Right Coronary Artery from the Pulmonary Artery: Long-Term Result After Takeuchi-Type Repair
6. Pollak JS, Saluja S, Thabet A, Henderson KJ, Denbow N, White Jr RI. Clinical and anatomic outcomes after embolotherapy of pulmonary arteriovenous malformations. J Vasc Interv Radiol 2006;17(1):35–44.
Case Reports
339
7. Faughnan ME, Thabet A, Mei-Zahav M, Colombo M, Maclusky I, Hyland RH, Pugash RA, Chait P, Henderson KJ, White RI. Pulmonary arteriovenous malformations in children: outcomes of transcatheter embolotherapy. J Pediatr 2004;145(6):826–31.
Aortopulmonary Window and Anomalous Origin of the Right Coronary Artery from the Pulmonary Artery: Long-Term Result After Takeuchi-Type Repair Davide Gabbieri, MD a , Mario Guadalupi, MD b and Guglielmo Stefanelli, MD a,∗ a
Department of Cardiovascular and Thoracic Surgery, Hesperia Hospital, Via Arqua` 80/A, 41100 Modena, Italy b Department of Cardiology, A. Perrino Hospital, Brindisi, Italy
We describe the case of a 2-month-old infant with the rare association of aortopulmonary window and anomalous origin of the right coronary artery from the pulmonary artery. The diagnosis was made by angiography and surgical repair was carried on employing the Takeuchi procedure. The angiographic control obtained 5 years after the operation showed an optimal surgical result. The latest echocardiographic follow-up, 24 years after the repair, has been fully satisfactory. (Heart, Lung and Circulation 2008;17:330–351) © 2007 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved. Keywords. Congenital heart disease (CHD); CHD; Great vessel anomalies; Heart failure
Introduction
A
nomalous origin of the right coronary artery (RCA) from the pulmonary artery (PA) in association with aortopulmonary window (APW) is an extremely rare cardiac anomaly. To our knowledge, only 15 cases of surgical correction of this abnormality have been published worldwide. This article reports the long-term result (24 years) of the surgical repair of this malformation and presents a literature review (Table 1).
Case Report In April 1982, a 3.5 kg 2-month-old boy was admitted to our Department with history of congestive heart failure (CHF). Cardiac catheterisation revealed the presence of an APW with the suspect of the origin of the RCA from the PA (Fig. 1A). The operation was carried on cardiopulmonary bypass with deep hypothermia. The RCA appeared to originate from the anterior aspect of the PA, 2 mm above the pulmonary valve. After cross-clamping of both the great vessels, cold crystalloid cardioplegia was injected into the Received 5 December 2006; received in revised form 22 January 2007; accepted 2 May 2007; available online 19 June 2007 ∗
Corresponding author. Tel.: +39 059449258; fax: +39 059449444. E-mail address: [email protected] (G. Stefanelli).
ascending aorta. A longitudinal pulmonary arteriotomy was performed, showing the oval-shaped, 6 mm × 10 mmsized, APW located few millimeters from the aortic valve plane. According to the Takeuchi technique,1 a tunnelshaped baffle of autologous pericardium was implanted inside the PA in order to connect the aorta to the anomalous coronary ostium through the APW. The PA was augmented with a second autologous pericardial patch. Operation and postoperative course were uneventful. The patient was discharged home on post-operative day 10 without long-term antiplatelets or anticoagulation therapy. Five years after the operation, a cardiac angiographic control revealed an optimal surgical result (Fig. 1B). At the last follow-up control, 24 years after surgery, the patient was in New York Heart Association functional class I without any restrictions of his active style of life. Transthoracic colour Doppler echocardiography showed a normal left and right ventricular ejection fraction, a right ventricle-PA mean gradient of 12 mmHg, and the RCA originating from the intrapulmonary channel (mean width, 5.6 mm) with normal flow (Fig. 2). An exercise test (cycle ergometer, James protocol) performed to evaluate the physical working capacity of the patient, was completely satisfactory (maximal power output: 1400 kg m/min, systolic pressure: 199 mmHg, diastolic pressure: 71 mmHg, heart rate: 196 beats/min). Furthermore, the exercise test ruled out the occurrence of any
© 2007 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved.
1443-9506/04/$30.00 doi:10.1016/j.hlc.2007.05.002
CASE REPORTS
Heart, Lung and Circulation 2008;17:330–351
CASE REPORTS
340 Case Reports
Table 1. Literature Review of Cases with APW and Anomalous Origin of the RCA from PA Author and References
Other Cardiac Anomalies
Shumacker Discussion of Cooley. Surgery 1957;42:101–20 Burroughs et al. J Cardiovasc Surg 1962;3:142–7 Morrow et al. Circulation 1962;25:463–76 Deverall et al. J Thorac Cardiovasc Surg 1969;57:479–86 Blieden et al. Br Heart J 1974;36:630–5 Luisi et al. Thorax 1980;35:446–8
None VSD GA malposition RAA VSD NS None
NS 16 years 7 years 10 months NS 4 months
Age
Langlois et al. Ann Chir 1980;34:216–23 Honek et al. Rozhl Chir 1984;63:749–53
NS SAS
NS 30 months
Casillas et al. Tex Heart Inst J 1986;13:325–31 Lloyd et al. Pediatr Cardiol 1987;8:153–4
None ALCAPA
3 months 10 months
Brouwer et al. Int J Cardiol 1990;28:384–6
None
4 months
Grunenfelder et al. Ann Thorac Surg 1999;67:233–5
ASD PDA
1 month
Izumoto et al. Ann Thorac Surg 1999;68:557–9
ASD VSD RAA
20 months
Greenway et al. Eur J Echocardiogr 2006;7:379–82
None
1.5 month, 3 months (2 pts.)
Surgery Partial APW ligature Coronary Ao connection and APW closure APW division suture Exploratory thoracotomy Transfer of the coronary ostium to the Ao Flap with RCA origin excised from PA and transferred to close APW NS Closure of APW with patch directing aortic blood into coronary ostia Intrapulmonary baffle Closure of APW with patch directing aortic blood into coronary ostia RCA excised from PA and transferred to Ao; closure of APW with patch RCA excised from PA as a button and transferred to Ao Closure of APW with patch directing aortic blood into coronary ostia RCA directly reimplanted into the Ao (both pts.)
Outcome Alive Dead Alive (RPS) Dead Dead Alive NS Alivea Alive Dead Alive Alive Alive Alive (both pts.)
ALCAPA: anomalous left coronary artery from pulmonary artery; Ao: aorta; APW: aortopulmonary window; ASD: atrial septal defect; CHF: congestive heart failure; GA: great arteries; NS: not specified; RCA: right coronary artery; PA: pulmonary artery; PDA: patent ductus arteriosus; pts.: patients; RAA: right aortic arch; RPS: residual pulmonary stenosis; SAS: subvalvular aortic stenosis; VSD: ventricular septal defect. a Long-term follow-up reported by S. Tuma et al. in Cesk Pediatr 1990;45:543–5.
Heart, Lung and Circulation 2008;17:330–351
Case Reports
341 CASE REPORTS
Heart, Lung and Circulation 2008;17:330–351
Figure 1. (A) Pre-operative angiogram showing simultaneous filling of aorta and pulmonary artery. (B) Angiographic control at 5 years showing the filling of right coronary artery from aorta.
Figure 2. Echocardiographic short-axis view: right coronary artery root inside pulmonary artery (arrow).
ischaemic electrocardiographic changes, confirming indirectly the absence of any limitation to the rise of the blood flow inside the intrapulmonary channel during the exercise.
Comment Aortopulmonary window is a communication between the main PA and the ascending aorta in the presence of two separate semilunar valves. Anomaly in the development of the spiral septum and alterations in the rotation of the outflow tracts occurring during the fifth and sixth weeks of the embryonic life cause this conotruncal cardiac anomaly.2 Isolated APW occurs in 0.2% of cases of congenital heart disease and is accompanied by other cardiac anomalies in 52% of cases. An anomalous origin of the coronary arteries from the PA represents the cardiovascular associated anomaly in the 5% of not-isolated APW.3 Therefore, the association between APW and anomalous coronary artery arising from the PA is an extremely rare cardiac malformation. Up to 1999, only 13 cases of the association of APW
and anomalous origin of the RCA from the PA and 10 cases of the association of APW and anomalous origin of the left coronary artery (LCA) from the PA were reported in literature.4–6 The patients present usually in a clinical status of CHF within the first months of life, reminding the clinical presentation of the Bland-White-Garland syndrome (anomalous origin of the LCA from the PA) associated with ventricular septal defect.4 The large left-to-right shunt is the main cause of cardiac failure and pulmonary hypertension. Electrocardiographic signs of myocardial ischaemia are not present, because the RCA or the LCA are filled with oxygenated blood streaming from the aorta into the PA through the APW.5 Although an accurate echocardiographic examination can be diagnostic and give all the informations required for a surgical repair,7,8 in selected cases an angiographic investigation can be necessary. However, preoperative angiographic diagnosis of anomalous origin of RCA or LCA associated with APW could be difficult because both great arteries fill simultaneously. Therefore, surgeons should be prepared to cope with coronary artery anomaly in the operative management of APW, although this association is extremely rare.6 The surgical correction of the association of APW and anomalous origin of the RCA from the PA necessarily include the connection of the anomalous RCA ostium with the aorta to avoid myocardial ischaemia. Different techniques were employed for such purpose, such as transfer of the coronary ostium to the aorta,9 rotation plastic procedure with pulmonary tissue patch,10 tunneling,11 and intrapulmonary baffling.4 This last surgical option, described in 1979 by Hamilton et al.12 and Takeuchi et al.1 for repair of anomalous left coronary artery from PA, was firstly employed by Casillas et al.4 in 1986 to correct an APW associated with anomalous origin of the RCA from PA. The anomalous coronary ostium was connected to the aorta using an intrapulmonary baffle of polytetrafluorethylene (PTFE) and the pulmonary arteriotomy was
342
Case Reports
CASE REPORTS
directly closed. In the present case, we also employed the Takeuchi procedure, but we used autologous pericardium in alternative to the PTFE, and preferred to augment the PA with a second patch to prevent residual pulmonary stenosis. Nowadays, the experience with arterial transfer in patients with transposition of the great arteries has made the trapdoor flap technique the modality standard for repair of the anomalous coronary artery, strongly reducing the role of other procedures.13 However, the Takeuchi technique could still represent an option in all those cases where the coronary relocation carries a higher risk of complications, i.e. when the coronary relocation would cause stretching or kinking of the artery or is impossible because the coronary take-off from the PA is far from the aorta. A long-term follow-up of the patients treated by the Takeuchi procedure is mandatory in order to exclude the potential late complications of this technique, namely obstruction of the tunnel itself, baffle leak creating a coronary–PA fistula, and varying degrees of supravalvular pulmonary stenosis.7 Non-invasive diagnostic techniques, such as transthoracic colour Doppler echocardiography and exercise test, could represent the first choice diagnostic tools to identify the patients requiring further invasive procedures, such as stress thallium scanning, positron emission tomographic scanning, and cardiac catheterisation with coronary angiography. To our knowledge, the follow-up time reported in this article, 24 years, is the longest available to verify the long-term result of the Takeuchi-type operation in this particular subset of malformations. Furthermore, from a surgical point of view, this long-term follow-up seems to confirm the importance of patch enlarging of the main PA when the Takeuchi technique is employed, to avoid a reoperation for supravalvular pulmonary stenosis.
Heart, Lung and Circulation 2008;17:330–351
2.
3. 4.
5.
6.
7.
8.
9. 10.
11.
12.
Acknowledgment The Authors would thank Miss Denise (Krealab, Modena, Italy) for her assistance in preparing the illustrations.
References 1. Takeuchi S, Imamura H, Katsumoto K, Hayashi I, Katohgi T, Yozu R, Ohkura M, Inoue T. New surgical method for repair
13.
of anomalous left coronary artery from pulmonary artery. J Thorac Cardiovasc Surg 1979;78:7–11. Webb S, Qayyum SR, Anderson RH, Lamers WH, Richardson MK. Septation and separation within the outflow tract of the developing heart. J Anat 2003;202:327–42. Kutsche LM, Van Mierop LH. Anatomy and pathogenesis of aorticopulmonary septal defect. Am J Cardiol 1987;59:443–7. Casillas JA, de Leon JP, Villagra F, Checa SL, Sanchez PA, Gomez R, Fortuny R, Brito JM. Aortopulmonary window with anomalous origin of the right coronary artery from the pulmonary trunk. Tex Heart Inst J 1986;13:325–31. Grunenfelder J, Zund G, Vogt PR, Turina MI. Aortopulmonary window with anomalous origin of the right coronary artery. Ann Thorac Surg 1999;67:233–5. Izumoto H, Ishihara K, Fujii Y, Oyama K, Kawazoe K. AP window and anomalous origin of right coronary artery from the window. Ann Thorac Surg 1999;68:557–9. Dodge-Khatami A, Mavroudis C, Backer CL. Anomalous origin of the left coronary artery from the pulmonary artery: collective review of surgical therapy. Ann Thorac Surg 2002;74:946–55. Greenway SC, Bradley TJ, Caldarone CA, Silverman NH, Hanley FL, Smallhorn JF. Aortopulmonary window with anomalous origin of the right coronary artery from the pulmonary artery: two cases highlighting the importance of complete pre-operative echocardiographic evaluation of the coronary arteries in all conotruncal anomalies. Eur J Echocardiogr 2006;7:379–82. Blieden LC, Moller JH. Aorticopulmonary septal defect. An experience with 17 patients. Br Heart J 1974;36:630–5. Luisi SV, Ashraf MH, Gula G, Radley-Smith R, Yacoub M. Anomalous origin of the right coronary artery with aortopulmonary window: functional and surgical considerations. Thorax 1980;35:446–8. Burroughs JT, Schmutzer KJ, Linder F, Neuhaus G. Anomalous origin of the right coronary artery with aorticopulmonary window and ventricular septal defect. Report of a case with complete operative correction. J Cardiovasc Surg (Torino) 1962;3:142–8. Hamilton DI, Ghosh PK, Donnelly RJ. An operation for anomalous origin of the left coronary artery. Br Heart J 1979;41:121–4. Ando M, Mee RB, Duncan BW, Drummond-Webb JJ, Seshadri SG, Mesia CI. Creation of a dual-coronary system for anomalous origin of the left coronary artery from the pulmonary artery utilizing the trapdoor flap technique. Eur J Cardiothorac Surg 2002;22:576–81.
Case Reports
339
7. Faughnan ME, Thabet A, Mei-Zahav M, Colombo M, Maclusky I, Hyland RH, Pugash RA, Chait P, Henderson KJ, White RI. Pulmonary arteriovenous malformations in children: outcomes of transcatheter embolotherapy. J Pediatr 2004;145(6):826–31.
Aortopulmonary Window and Anomalous Origin of the Right Coronary Artery from the Pulmonary Artery: Long-Term Result After Takeuchi-Type Repair Davide Gabbieri, MD a , Mario Guadalupi, MD b and Guglielmo Stefanelli, MD a,∗ a
Department of Cardiovascular and Thoracic Surgery, Hesperia Hospital, Via Arqua` 80/A, 41100 Modena, Italy b Department of Cardiology, A. Perrino Hospital, Brindisi, Italy
We describe the case of a 2-month-old infant with the rare association of aortopulmonary window and anomalous origin of the right coronary artery from the pulmonary artery. The diagnosis was made by angiography and surgical repair was carried on employing the Takeuchi procedure. The angiographic control obtained 5 years after the operation showed an optimal surgical result. The latest echocardiographic follow-up, 24 years after the repair, has been fully satisfactory. (Heart, Lung and Circulation 2008;17:330–351) © 2007 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved. Keywords. Congenital heart disease (CHD); CHD; Great vessel anomalies; Heart failure
Introduction
A
nomalous origin of the right coronary artery (RCA) from the pulmonary artery (PA) in association with aortopulmonary window (APW) is an extremely rare cardiac anomaly. To our knowledge, only 15 cases of surgical correction of this abnormality have been published worldwide. This article reports the long-term result (24 years) of the surgical repair of this malformation and presents a literature review (Table 1).
Case Report In April 1982, a 3.5 kg 2-month-old boy was admitted to our Department with history of congestive heart failure (CHF). Cardiac catheterisation revealed the presence of an APW with the suspect of the origin of the RCA from the PA (Fig. 1A). The operation was carried on cardiopulmonary bypass with deep hypothermia. The RCA appeared to originate from the anterior aspect of the PA, 2 mm above the pulmonary valve. After cross-clamping of both the great vessels, cold crystalloid cardioplegia was injected into the Received 5 December 2006; received in revised form 22 January 2007; accepted 2 May 2007; available online 19 June 2007 ∗
Corresponding author. Tel.: +39 059449258; fax: +39 059449444. E-mail address: [email protected] (G. Stefanelli).
ascending aorta. A longitudinal pulmonary arteriotomy was performed, showing the oval-shaped, 6 mm × 10 mmsized, APW located few millimeters from the aortic valve plane. According to the Takeuchi technique,1 a tunnelshaped baffle of autologous pericardium was implanted inside the PA in order to connect the aorta to the anomalous coronary ostium through the APW. The PA was augmented with a second autologous pericardial patch. Operation and postoperative course were uneventful. The patient was discharged home on post-operative day 10 without long-term antiplatelets or anticoagulation therapy. Five years after the operation, a cardiac angiographic control revealed an optimal surgical result (Fig. 1B). At the last follow-up control, 24 years after surgery, the patient was in New York Heart Association functional class I without any restrictions of his active style of life. Transthoracic colour Doppler echocardiography showed a normal left and right ventricular ejection fraction, a right ventricle-PA mean gradient of 12 mmHg, and the RCA originating from the intrapulmonary channel (mean width, 5.6 mm) with normal flow (Fig. 2). An exercise test (cycle ergometer, James protocol) performed to evaluate the physical working capacity of the patient, was completely satisfactory (maximal power output: 1400 kg m/min, systolic pressure: 199 mmHg, diastolic pressure: 71 mmHg, heart rate: 196 beats/min). Furthermore, the exercise test ruled out the occurrence of any
© 2007 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved.
1443-9506/04/$30.00 doi:10.1016/j.hlc.2007.05.002
CASE REPORTS
Heart, Lung and Circulation 2008;17:330–351
CASE REPORTS
340 Case Reports
Table 1. Literature Review of Cases with APW and Anomalous Origin of the RCA from PA Author and References
Other Cardiac Anomalies
Shumacker Discussion of Cooley. Surgery 1957;42:101–20 Burroughs et al. J Cardiovasc Surg 1962;3:142–7 Morrow et al. Circulation 1962;25:463–76 Deverall et al. J Thorac Cardiovasc Surg 1969;57:479–86 Blieden et al. Br Heart J 1974;36:630–5 Luisi et al. Thorax 1980;35:446–8
None VSD GA malposition RAA VSD NS None
NS 16 years 7 years 10 months NS 4 months
Age
Langlois et al. Ann Chir 1980;34:216–23 Honek et al. Rozhl Chir 1984;63:749–53
NS SAS
NS 30 months
Casillas et al. Tex Heart Inst J 1986;13:325–31 Lloyd et al. Pediatr Cardiol 1987;8:153–4
None ALCAPA
3 months 10 months
Brouwer et al. Int J Cardiol 1990;28:384–6
None
4 months
Grunenfelder et al. Ann Thorac Surg 1999;67:233–5
ASD PDA
1 month
Izumoto et al. Ann Thorac Surg 1999;68:557–9
ASD VSD RAA
20 months
Greenway et al. Eur J Echocardiogr 2006;7:379–82
None
1.5 month, 3 months (2 pts.)
Surgery Partial APW ligature Coronary Ao connection and APW closure APW division suture Exploratory thoracotomy Transfer of the coronary ostium to the Ao Flap with RCA origin excised from PA and transferred to close APW NS Closure of APW with patch directing aortic blood into coronary ostia Intrapulmonary baffle Closure of APW with patch directing aortic blood into coronary ostia RCA excised from PA and transferred to Ao; closure of APW with patch RCA excised from PA as a button and transferred to Ao Closure of APW with patch directing aortic blood into coronary ostia RCA directly reimplanted into the Ao (both pts.)
Outcome Alive Dead Alive (RPS) Dead Dead Alive NS Alivea Alive Dead Alive Alive Alive Alive (both pts.)
ALCAPA: anomalous left coronary artery from pulmonary artery; Ao: aorta; APW: aortopulmonary window; ASD: atrial septal defect; CHF: congestive heart failure; GA: great arteries; NS: not specified; RCA: right coronary artery; PA: pulmonary artery; PDA: patent ductus arteriosus; pts.: patients; RAA: right aortic arch; RPS: residual pulmonary stenosis; SAS: subvalvular aortic stenosis; VSD: ventricular septal defect. a Long-term follow-up reported by S. Tuma et al. in Cesk Pediatr 1990;45:543–5.
Heart, Lung and Circulation 2008;17:330–351
Case Reports
341 CASE REPORTS
Heart, Lung and Circulation 2008;17:330–351
Figure 1. (A) Pre-operative angiogram showing simultaneous filling of aorta and pulmonary artery. (B) Angiographic control at 5 years showing the filling of right coronary artery from aorta.
Figure 2. Echocardiographic short-axis view: right coronary artery root inside pulmonary artery (arrow).
ischaemic electrocardiographic changes, confirming indirectly the absence of any limitation to the rise of the blood flow inside the intrapulmonary channel during the exercise.
Comment Aortopulmonary window is a communication between the main PA and the ascending aorta in the presence of two separate semilunar valves. Anomaly in the development of the spiral septum and alterations in the rotation of the outflow tracts occurring during the fifth and sixth weeks of the embryonic life cause this conotruncal cardiac anomaly.2 Isolated APW occurs in 0.2% of cases of congenital heart disease and is accompanied by other cardiac anomalies in 52% of cases. An anomalous origin of the coronary arteries from the PA represents the cardiovascular associated anomaly in the 5% of not-isolated APW.3 Therefore, the association between APW and anomalous coronary artery arising from the PA is an extremely rare cardiac malformation. Up to 1999, only 13 cases of the association of APW
and anomalous origin of the RCA from the PA and 10 cases of the association of APW and anomalous origin of the left coronary artery (LCA) from the PA were reported in literature.4–6 The patients present usually in a clinical status of CHF within the first months of life, reminding the clinical presentation of the Bland-White-Garland syndrome (anomalous origin of the LCA from the PA) associated with ventricular septal defect.4 The large left-to-right shunt is the main cause of cardiac failure and pulmonary hypertension. Electrocardiographic signs of myocardial ischaemia are not present, because the RCA or the LCA are filled with oxygenated blood streaming from the aorta into the PA through the APW.5 Although an accurate echocardiographic examination can be diagnostic and give all the informations required for a surgical repair,7,8 in selected cases an angiographic investigation can be necessary. However, preoperative angiographic diagnosis of anomalous origin of RCA or LCA associated with APW could be difficult because both great arteries fill simultaneously. Therefore, surgeons should be prepared to cope with coronary artery anomaly in the operative management of APW, although this association is extremely rare.6 The surgical correction of the association of APW and anomalous origin of the RCA from the PA necessarily include the connection of the anomalous RCA ostium with the aorta to avoid myocardial ischaemia. Different techniques were employed for such purpose, such as transfer of the coronary ostium to the aorta,9 rotation plastic procedure with pulmonary tissue patch,10 tunneling,11 and intrapulmonary baffling.4 This last surgical option, described in 1979 by Hamilton et al.12 and Takeuchi et al.1 for repair of anomalous left coronary artery from PA, was firstly employed by Casillas et al.4 in 1986 to correct an APW associated with anomalous origin of the RCA from PA. The anomalous coronary ostium was connected to the aorta using an intrapulmonary baffle of polytetrafluorethylene (PTFE) and the pulmonary arteriotomy was
342
Case Reports
CASE REPORTS
directly closed. In the present case, we also employed the Takeuchi procedure, but we used autologous pericardium in alternative to the PTFE, and preferred to augment the PA with a second patch to prevent residual pulmonary stenosis. Nowadays, the experience with arterial transfer in patients with transposition of the great arteries has made the trapdoor flap technique the modality standard for repair of the anomalous coronary artery, strongly reducing the role of other procedures.13 However, the Takeuchi technique could still represent an option in all those cases where the coronary relocation carries a higher risk of complications, i.e. when the coronary relocation would cause stretching or kinking of the artery or is impossible because the coronary take-off from the PA is far from the aorta. A long-term follow-up of the patients treated by the Takeuchi procedure is mandatory in order to exclude the potential late complications of this technique, namely obstruction of the tunnel itself, baffle leak creating a coronary–PA fistula, and varying degrees of supravalvular pulmonary stenosis.7 Non-invasive diagnostic techniques, such as transthoracic colour Doppler echocardiography and exercise test, could represent the first choice diagnostic tools to identify the patients requiring further invasive procedures, such as stress thallium scanning, positron emission tomographic scanning, and cardiac catheterisation with coronary angiography. To our knowledge, the follow-up time reported in this article, 24 years, is the longest available to verify the long-term result of the Takeuchi-type operation in this particular subset of malformations. Furthermore, from a surgical point of view, this long-term follow-up seems to confirm the importance of patch enlarging of the main PA when the Takeuchi technique is employed, to avoid a reoperation for supravalvular pulmonary stenosis.
Heart, Lung and Circulation 2008;17:330–351
2.
3. 4.
5.
6.
7.
8.
9. 10.
11.
12.
Acknowledgment The Authors would thank Miss Denise (Krealab, Modena, Italy) for her assistance in preparing the illustrations.
References 1. Takeuchi S, Imamura H, Katsumoto K, Hayashi I, Katohgi T, Yozu R, Ohkura M, Inoue T. New surgical method for repair
13.
of anomalous left coronary artery from pulmonary artery. J Thorac Cardiovasc Surg 1979;78:7–11. Webb S, Qayyum SR, Anderson RH, Lamers WH, Richardson MK. Septation and separation within the outflow tract of the developing heart. J Anat 2003;202:327–42. Kutsche LM, Van Mierop LH. Anatomy and pathogenesis of aorticopulmonary septal defect. Am J Cardiol 1987;59:443–7. Casillas JA, de Leon JP, Villagra F, Checa SL, Sanchez PA, Gomez R, Fortuny R, Brito JM. Aortopulmonary window with anomalous origin of the right coronary artery from the pulmonary trunk. Tex Heart Inst J 1986;13:325–31. Grunenfelder J, Zund G, Vogt PR, Turina MI. Aortopulmonary window with anomalous origin of the right coronary artery. Ann Thorac Surg 1999;67:233–5. Izumoto H, Ishihara K, Fujii Y, Oyama K, Kawazoe K. AP window and anomalous origin of right coronary artery from the window. Ann Thorac Surg 1999;68:557–9. Dodge-Khatami A, Mavroudis C, Backer CL. Anomalous origin of the left coronary artery from the pulmonary artery: collective review of surgical therapy. Ann Thorac Surg 2002;74:946–55. Greenway SC, Bradley TJ, Caldarone CA, Silverman NH, Hanley FL, Smallhorn JF. Aortopulmonary window with anomalous origin of the right coronary artery from the pulmonary artery: two cases highlighting the importance of complete pre-operative echocardiographic evaluation of the coronary arteries in all conotruncal anomalies. Eur J Echocardiogr 2006;7:379–82. Blieden LC, Moller JH. Aorticopulmonary septal defect. An experience with 17 patients. Br Heart J 1974;36:630–5. Luisi SV, Ashraf MH, Gula G, Radley-Smith R, Yacoub M. Anomalous origin of the right coronary artery with aortopulmonary window: functional and surgical considerations. Thorax 1980;35:446–8. Burroughs JT, Schmutzer KJ, Linder F, Neuhaus G. Anomalous origin of the right coronary artery with aorticopulmonary window and ventricular septal defect. Report of a case with complete operative correction. J Cardiovasc Surg (Torino) 1962;3:142–8. Hamilton DI, Ghosh PK, Donnelly RJ. An operation for anomalous origin of the left coronary artery. Br Heart J 1979;41:121–4. Ando M, Mee RB, Duncan BW, Drummond-Webb JJ, Seshadri SG, Mesia CI. Creation of a dual-coronary system for anomalous origin of the left coronary artery from the pulmonary artery utilizing the trapdoor flap technique. Eur J Cardiothorac Surg 2002;22:576–81.