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Transcatheter closure for baffle leak after Takeuchi repair of anomalous left coronary artery from the pulmonary artery: a case report

Transcatheter closure for baffle leak after Takeuchi repair of anomalous left coronary artery... CASE REPORT European Heart Journal - Case Reports (2018) 2, 1–4 doi:10.1093/ehjcr/yty028 Congenital heart disease Transcatheter closure for baffle leak after Takeuchi repair of anomalous left coronary artery from the pulmonary artery: a case report Hu Hai-Bo*, Yang Kai, and Pan Xiang-bin Center of Structural Heart Disease, Fuwai Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100037, China Received 17 November 2017; accepted 11 February 2018; online publish-ahead-of-print 29 March 2018 Introduction Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare but serious congenital coronary abnormality, Takeuchi repair is an alternative treatment option for this anomaly in certain situations, it is reported that baffle leak is the most common complication after Takeuchi repair, and some of this complication re- quire surgical reoperation. ................................................................................................................................................................................................... Case In this case, a 43-year-old woman diagnosed with ALCAPA underwent Takeuchi procedure 6 months ago, presentation 4 months after the procedure, the patient complained of chest pain on exertion and was confirmed to have baffle leak, and then we treated this complication successfully by percutaneous transcatheter closure. ................................................................................................................................................................................................... Discussion Baffle leak is the most common complication after Takeuchi procedure of ALCAPA, some of them require surgical reoperation. In this case, we introduce a new method, percutaneous transcatheter closure, to treat the baffle leak. To our knowledge, this is the first reported case of transcatheter closure for baffle leak after Takeuchi repair, which may be an alternative treatment option for the baffle leak after Takeuchi repair of this rare congenital coron- ary anomaly. Keywords Case report Coronary anomaly ALCAPA Takeuchi Baffle leak Transcatheter closure • • • • • Introduction Learning points Anomalous origin of the left coronary artery from the pulmonary ar- • . Baffle leak is the most common complication after Takeuchi tery (ALCAPA) is a rare but serious congenital coronary abnormality procedure of anomalous origin of the left coronary artery that if left untreated, results in high mortality. There are two types of from the pulmonary artery. In this case, we introduce a new ALCAPA: the infant type and the adult type, each of which has differ- method, percutaneous transcatheter closure, to treat the baffle . ent clinical manifestations and outcomes, which were described in leak, which is an alternative treatment option for the baffle . . 2 . detail by Pe~ na et al. Once ALCAPA is diagnosed, early surgical treat- leak and may be useful to clinical and interventional cardiolo- . . ment is vital to correct the anomaly and prevent complications. The gists who care for patients with structural heart disease. . preferred surgical method is restoring a two-coronary-artery circula- The interventional procedure should be very soft and careful . . 1,2 . tion system, and Takeuchi repair is an alternative treatment to avoid the injury and occlusion of the coronary after the im- . . 1,3 . method for ALCAPA in certain situations. Late complications after plantation of the device. Takeuchi repair of ALCAPA were reviewed by Ginde et al., and it is * Corresponding author. Tel: 15600300802, Email: fwyyykai@sina.com or hhb1999@sina.com. This case report was reviewed by George Giannakoulas and Ina Michel-Behnke. V The Author(s) 2018. Published by Oxford University Press on behalf of the European Society of Cardiology. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/2/1/4956396 by Ed 'DeepDyve' Gillespie user on 03 July 2018 2 H. Hai-Bo et al. reported that baffle leak is the most common complication. Since border between the 2nd and 4th rib, blood pressure (BP) 110/ some of this complication require reoperation, we are going to intro- 54 mmHg, heart rate 75 b.p.m. Electrocardiogram showed left ven- duce a new treatment method for the baffle leak and our experience . tricle high voltage and abnormal Q waves. Transthoracic echocardio- of percutaneous transcatheter closure. . gram (TTE) revealed a small fistula from the intrapulmonary baffle to . the main pulmonary artery (MPA), mild mitral regurgitation, left ven- . tricular end-diastolic diameter (LVEDD) 54 mm (57 mm before the Timeline . Takeuchi repair), and ejection fraction 52%. Cardiac computed tom- . ography angiography (CTA) confirmed a fistulous connection from the posterior wall of the proximal LCA into the anterior wall of MPA, the diameter of the fistula is about 4 mm (see Figure 2A). After the baffle leak was confirmed, we adopted a new method, Time Events ................................................................................................. . percutaneous transcatheter closure, to treat the baffle leak. During the operation, ascending aortic angiography in the left anterior ob- 20 years ago Chest pain occasionally, the patient didn’t lique (LAO) view and selective coronary angiography of the left pay much attention to it and no special main coronary artery demonstrated contrast leakage from the LCA treatment was obtained into the MPA (see Figure 3A and B), then we used a ventricular septal 6 months ago The patient came to our hospital due to defect (VSD) occluder (Starway Medical Technology Inc., Beijing, worsening symptoms and was diagnosed China) to close the baffle leak through 6 Fr transferring sheath, the with anomalous origin of the left coron- VSD occluder we used is a self-expandable, double disc implant de- ary artery from the pulmonary artery and . vice made from a Nitinol wire mesh, the two discs are linked to- treated with Takeuchi procedure . gether by a short connecting waist, and the device waist in diameter Within 3 months No abnormality . is 6 mm. Selective coronary angiography of the left main coronary ar- after the Takeuchi . tery in the LAO view demonstrated no contrast leakage after im- repair . plantation of the device (see Figures 2B and 3C). Blood pressure 4 months after the Chief complaint: chest pain on exertion . . increased to 115/68 mmHg. The pulmonary artery pressure (PAP) Takeuchi repair Physical examination: Grade 3/6 continuous . decreased from 46/22 (mean: 29) mmHg to 31/14 (mean: 19) murmur at left sternal border between . mmHg. The Q /Q decreased from 1.52 to 1.05. Twenty-four hours the 2nd and 4th rib p s . after the interventional treatment, TTE re-examination showed no Imaging: a small fistula from the intrapulmo- . evident residual shunt and LVEDD decreased from 54 mm to nary baffle to the main pulmonary artery . 50 mm. The patient’s symptom improved significantly before hos- Treatment: percutaneous transcatheter pital discharge, and TTE re-examination of 3-month follow-up closure showed no evident residual shunt. 3 months after the No evident residual shunt was observed transcatheter closure . Discussion Anomalous origin of the left coronary artery from the pulmonary ar- Case presentation tery (ALCAPA) is a rare but serious congenital heart disease and re- Twenty years ago, a young woman (23 years old) felt chest pain occa- quires surgical repairs once ALCAPA is diagnosed, Takeuchi repair is sionally when she walked hastily, the pain lasted about 2 min and can an alternative treatment option for this anomaly in certain situations, 1,3–7 relieve itself, so she was admitted to the local hospital and diagnosed although there is some controversy about this procedure. It was with mild-moderate mitral insufficiency (MI). Because of no obvious first described in 1979, which consists of using a flap derived from limitation in physical activity, the patient didn’t pay much attention to the anterior wall of the pulmonary trunk to create an internal tunnel it, and no special treatment was obtained. Six months ago, she came in the main pulmonary trunk between a surgically created aortopul- to Beijing for further diagnosis due to worsening symptoms, she was monary window and the left coronary ostium, the opening in the 1,8,9 diagnosed with congenital heart disease, ALCAPA (see Figure 1), se- pulmonary trunk is patched with pericardium. Reported compli- vere MI, and left ventricular enlargement (LVE) and underwent . cations of Takeuchi repair mainly include baffle leak and supravalvular Takeuchi procedure and concomitant mitral valve repair in our hos- . pulmonary stenosis, and the baffle leak is the most common compli- . 1,4 pital. During the operation, we found that the left coronary artery . cation (27%). We reviewed related articles in recent 20 years and (LCA) arises from the left sinus of pulmonary trunk, and we created . summarized some information about the baffle leak after Takeuchi an aortopulmonary window above the left aortic sinus and an intra- . repair of ALCAPA (see Table 1). It shows that the incidence rate of pulmonary tunnel that baffles the aorta to the ostium of the anomal- . baffle leak is about 27.8%, which is similar to the study by Ginde ous LCA through the anterior wall of the pulmonary trunk. After the et al., and 9.7% of patients received surgical reoperation. operation, she was followed regularly for 3 months and unaware of In this case, the patient was confirmed to have baffle leak 4 any abnormalities. Four months after the Takeuchi repair, she began months after the Takeuchi repair, and the diameter of the fistula to complain of chest pain on exertion. Physical examination revealed was about 4 mm, which was likely to explain the reason why the a Grade 3/6 continuous murmur of puffing character at left sternal patient felt chest pain on exertion. In addition to the patient’s Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/2/1/4956396 by Ed 'DeepDyve' Gillespie user on 03 July 2018 Transcatheter closure for baffle leak 3 Figure 1 Before the Takeuchi repair, computed tomography angiography shows that the left coronary artery arises from the main pulmonary artery. Figure 2 Four months after the Takeuchi repair, computed tomography angiography confirms a fistulous connection from the baffle into the main pulmonary artery, the diameter of the fistula is about 4 mm (A, red arrow); after the interventional therapy, the red arrow shows the device (B). symptom, this complication may result in certain sequelae if un- 54 mmHg to 68 mmHg, which means improved coronary perfusion treated, such as chronic myocardial ischaemia and angina, myocar- after the baffle leak closure, and PAP decreased due to shunt clos- dial infarction, heart failure, pulmonary hypertension, endocarditis, ure and probably improved cardiac function. To our knowledge, and so on, therefore the patient had a strong will to be treated this is the first reported case of transcatheter closure for baffle after the baffle leak was confirmed. Because of the short time after leak after Takeuchi repair. There are still no available relevant art- the first thoracotomy (only 4 months) and the difficulty, severe icles at home and abroad we can learn from. Our initial experience trauma and high risks of second thoracotomy, such as post-opera- is that: (i) make clear the relationship between the fistula and the tive adhesion, bleeding, respiratory failure, pulmonary infection, ostium of coronary, evaluate the feasibility of percutaneous trans- pulmonary atelectasis, pleural effusion, and so on, we finally catheter closure, (ii) interventional procedure should be very soft reached an agreement with cardiac surgeons and decided to treat to avoid the injury of coronary, (iii) avoid the occlusion of coronary the baffle leak through percutaneous transcatheter closure, which . after the implantation of occluder. But there are still some prob- has the advantages of lesser trauma and haemorrhage, slight pain, . lems have to be addressed, for example, what kind of baffle leak and rapid recovery. The patient was informed and willing to be . can be treated by percutaneous transcatheter closure? What type treated by this method. After the interventional therapy, the pa- . of occluder can be used for baffle leak? What size of occluder tient’s symptom improved significantly, diastolic BP increased from . should be recommended? Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/2/1/4956396 by Ed 'DeepDyve' Gillespie user on 03 July 2018 4 H. Hai-Bo et al. Figure 3 Percutaneous transcatheter closure: ascending aortic angiography in the left anterior oblique view (A) and selective coronary angiography of the left main coronary artery (B) demonstrates contrast leakage from the left coronary artery into the main pulmonary artery (red arrow); select- ive coronary angiography of the left main coronary artery in the left anterior oblique view (C) demonstrates no contrast leakage after implantation of the device (red arrow). associated text has been obtained from the patient in line with COPE Table 1 Summary of studies on baffle leak after guidance. Takeuchi repair of anomalous origin of the left coronary . artery from the pulmonary artery Conflict of interest: none declared. Study Year No. of Baffle Reoperation References patients leak . ................................................................................................. . 1. Ginde S, Earing MG, Bartz PJ, Cava JR, Tweddell JS. Late complications after 4 . Takeuchi repair of anomalous left coronary artery from the pulmonary artery: Schwartz et al. 1997 21 11 3 . case series and review of literature. Pediatr Cardiol 2012;33:1115–1123. 3 . Cochrane et al. 1998 12 2 0 . 2. Pe~ na E, Nguyen ET, Merchant N, Dennie C. ALCAPA syndrome: not just a pedi- 10 . Birk et al. 2000 7 1 1 . atric disease. RadioGraphics 2009;29:553–565. 3. Cochrane AD, Coleman DM, Davis AM, Brizard CP, Wolfe R, Karl TR. Excellent Michielon et al. 2003 12 1 1 long-term functional outcome after an operation for anomalous left coronary ar- Ojala et al. 2009 4 1 0 . tery from the pulmonary artery. J Thorac Cardiovasc Surg 1999;117:332–342. Ginde et al. 2012 9 3 1 4. Schwartz ML, Jonas RA, Colan SD. Anomalous origin of left coronary artery from pulmonary artery: recovery of left ventricular function after dual coronary Quanlin and Jie 2013 7 1 1 repair. J Am Coll Cardiol 1997;30:547–553. Total (%) 72 20 (27.8) 7 (9.7) . 5. Michielon G, Di Carlo D, Brancaccio G, Guccione P, Mazzera E, Toscano A, Di . Donato RM. Anomalous coronary artery origin from the pulmonary artery: cor- relation between surgical timing and left ventricular function recovery. Ann Thorac Surg 2003;76:581–588. . 6. Ojala T, Salminen J, Happonen J-M, Pihkala J, Jokinen E, Sairanen H. Excellent functional result in children after correction of anomalous origin of left coronary Conclusion artery from the pulmonary artery—a population-based complete follow-up study. Interact Cardiovasc Thorac Surg 2010;10:70–75. Percutaneous transcatheter closure is an alternative treatment option . 7. Yu-Juan Q, Pei-Jun L. Advancement of surgical therapy for anomalous left coron- for the baffle leak after Takeuchi repair of ALCAPA, this case is just an . ary artery from pulmonary artery. Chin J Clin Thorac Cardiovasc Surg 2017;24: 239–242. initial exploration, more experience, and clinical data are still needed. . . 8. Takeuchi S, Imamura H, Katsumoto K, Hayashi I, Katohgi T, Yozu R, Ohkura M, Inoue T. New surgical method for repair of anomalous left coronary artery from pulmonary artery. J Thorac Cardiovasc Surg 1979;78:7–11. 9. Dua R, Smith JA, Wilkinson JL, Menahem S, Karl TR, Goh TH, Mee RBB. Long- Supplementary material . term follow-up after two coronary repair of anomalous left coronary artery from the pulmonary artery. J Cardiac Surg 1993;8:384–390. Supplementary material is available at European Heart Journal – Case 10. Birk E, Stamler A, Katz J, Berant M, Dagan O, Matitiau A, Erez E, Blieden LC, . Vidne BA. Anomalous origin of the left coronary artery from the pulmonary ar- Reports online. tery: diagnosis and postoperative follow up. Isr Med Assoc J 2000;2:111–114. 11. Quanlin M, Jie X. Surgical treatment of anomalous origin of the left coronary Consent: The author/s confirm that written consent for submis- artery from the pulmonary artery. Chin J Ethnomed Ethnopharm 2013;15: . 42–43. sion and publication of this case report including image(s) and Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/2/1/4956396 by Ed 'DeepDyve' Gillespie user on 03 July 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png European Heart Journal - Case Reports Oxford University Press

Transcatheter closure for baffle leak after Takeuchi repair of anomalous left coronary artery from the pulmonary artery: a case report

European Heart Journal - Case Reports , Volume Advance Article (2) – Mar 29, 2018

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© The Author(s) 2018. Published by Oxford University Press on behalf of the European Society of Cardiology.
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10.1093/ehjcr/yty028
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Abstract

CASE REPORT European Heart Journal - Case Reports (2018) 2, 1–4 doi:10.1093/ehjcr/yty028 Congenital heart disease Transcatheter closure for baffle leak after Takeuchi repair of anomalous left coronary artery from the pulmonary artery: a case report Hu Hai-Bo*, Yang Kai, and Pan Xiang-bin Center of Structural Heart Disease, Fuwai Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100037, China Received 17 November 2017; accepted 11 February 2018; online publish-ahead-of-print 29 March 2018 Introduction Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) is a rare but serious congenital coronary abnormality, Takeuchi repair is an alternative treatment option for this anomaly in certain situations, it is reported that baffle leak is the most common complication after Takeuchi repair, and some of this complication re- quire surgical reoperation. ................................................................................................................................................................................................... Case In this case, a 43-year-old woman diagnosed with ALCAPA underwent Takeuchi procedure 6 months ago, presentation 4 months after the procedure, the patient complained of chest pain on exertion and was confirmed to have baffle leak, and then we treated this complication successfully by percutaneous transcatheter closure. ................................................................................................................................................................................................... Discussion Baffle leak is the most common complication after Takeuchi procedure of ALCAPA, some of them require surgical reoperation. In this case, we introduce a new method, percutaneous transcatheter closure, to treat the baffle leak. To our knowledge, this is the first reported case of transcatheter closure for baffle leak after Takeuchi repair, which may be an alternative treatment option for the baffle leak after Takeuchi repair of this rare congenital coron- ary anomaly. Keywords Case report Coronary anomaly ALCAPA Takeuchi Baffle leak Transcatheter closure • • • • • Introduction Learning points Anomalous origin of the left coronary artery from the pulmonary ar- • . Baffle leak is the most common complication after Takeuchi tery (ALCAPA) is a rare but serious congenital coronary abnormality procedure of anomalous origin of the left coronary artery that if left untreated, results in high mortality. There are two types of from the pulmonary artery. In this case, we introduce a new ALCAPA: the infant type and the adult type, each of which has differ- method, percutaneous transcatheter closure, to treat the baffle . ent clinical manifestations and outcomes, which were described in leak, which is an alternative treatment option for the baffle . . 2 . detail by Pe~ na et al. Once ALCAPA is diagnosed, early surgical treat- leak and may be useful to clinical and interventional cardiolo- . . ment is vital to correct the anomaly and prevent complications. The gists who care for patients with structural heart disease. . preferred surgical method is restoring a two-coronary-artery circula- The interventional procedure should be very soft and careful . . 1,2 . tion system, and Takeuchi repair is an alternative treatment to avoid the injury and occlusion of the coronary after the im- . . 1,3 . method for ALCAPA in certain situations. Late complications after plantation of the device. Takeuchi repair of ALCAPA were reviewed by Ginde et al., and it is * Corresponding author. Tel: 15600300802, Email: fwyyykai@sina.com or hhb1999@sina.com. This case report was reviewed by George Giannakoulas and Ina Michel-Behnke. V The Author(s) 2018. Published by Oxford University Press on behalf of the European Society of Cardiology. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/2/1/4956396 by Ed 'DeepDyve' Gillespie user on 03 July 2018 2 H. Hai-Bo et al. reported that baffle leak is the most common complication. Since border between the 2nd and 4th rib, blood pressure (BP) 110/ some of this complication require reoperation, we are going to intro- 54 mmHg, heart rate 75 b.p.m. Electrocardiogram showed left ven- duce a new treatment method for the baffle leak and our experience . tricle high voltage and abnormal Q waves. Transthoracic echocardio- of percutaneous transcatheter closure. . gram (TTE) revealed a small fistula from the intrapulmonary baffle to . the main pulmonary artery (MPA), mild mitral regurgitation, left ven- . tricular end-diastolic diameter (LVEDD) 54 mm (57 mm before the Timeline . Takeuchi repair), and ejection fraction 52%. Cardiac computed tom- . ography angiography (CTA) confirmed a fistulous connection from the posterior wall of the proximal LCA into the anterior wall of MPA, the diameter of the fistula is about 4 mm (see Figure 2A). After the baffle leak was confirmed, we adopted a new method, Time Events ................................................................................................. . percutaneous transcatheter closure, to treat the baffle leak. During the operation, ascending aortic angiography in the left anterior ob- 20 years ago Chest pain occasionally, the patient didn’t lique (LAO) view and selective coronary angiography of the left pay much attention to it and no special main coronary artery demonstrated contrast leakage from the LCA treatment was obtained into the MPA (see Figure 3A and B), then we used a ventricular septal 6 months ago The patient came to our hospital due to defect (VSD) occluder (Starway Medical Technology Inc., Beijing, worsening symptoms and was diagnosed China) to close the baffle leak through 6 Fr transferring sheath, the with anomalous origin of the left coron- VSD occluder we used is a self-expandable, double disc implant de- ary artery from the pulmonary artery and . vice made from a Nitinol wire mesh, the two discs are linked to- treated with Takeuchi procedure . gether by a short connecting waist, and the device waist in diameter Within 3 months No abnormality . is 6 mm. Selective coronary angiography of the left main coronary ar- after the Takeuchi . tery in the LAO view demonstrated no contrast leakage after im- repair . plantation of the device (see Figures 2B and 3C). Blood pressure 4 months after the Chief complaint: chest pain on exertion . . increased to 115/68 mmHg. The pulmonary artery pressure (PAP) Takeuchi repair Physical examination: Grade 3/6 continuous . decreased from 46/22 (mean: 29) mmHg to 31/14 (mean: 19) murmur at left sternal border between . mmHg. The Q /Q decreased from 1.52 to 1.05. Twenty-four hours the 2nd and 4th rib p s . after the interventional treatment, TTE re-examination showed no Imaging: a small fistula from the intrapulmo- . evident residual shunt and LVEDD decreased from 54 mm to nary baffle to the main pulmonary artery . 50 mm. The patient’s symptom improved significantly before hos- Treatment: percutaneous transcatheter pital discharge, and TTE re-examination of 3-month follow-up closure showed no evident residual shunt. 3 months after the No evident residual shunt was observed transcatheter closure . Discussion Anomalous origin of the left coronary artery from the pulmonary ar- Case presentation tery (ALCAPA) is a rare but serious congenital heart disease and re- Twenty years ago, a young woman (23 years old) felt chest pain occa- quires surgical repairs once ALCAPA is diagnosed, Takeuchi repair is sionally when she walked hastily, the pain lasted about 2 min and can an alternative treatment option for this anomaly in certain situations, 1,3–7 relieve itself, so she was admitted to the local hospital and diagnosed although there is some controversy about this procedure. It was with mild-moderate mitral insufficiency (MI). Because of no obvious first described in 1979, which consists of using a flap derived from limitation in physical activity, the patient didn’t pay much attention to the anterior wall of the pulmonary trunk to create an internal tunnel it, and no special treatment was obtained. Six months ago, she came in the main pulmonary trunk between a surgically created aortopul- to Beijing for further diagnosis due to worsening symptoms, she was monary window and the left coronary ostium, the opening in the 1,8,9 diagnosed with congenital heart disease, ALCAPA (see Figure 1), se- pulmonary trunk is patched with pericardium. Reported compli- vere MI, and left ventricular enlargement (LVE) and underwent . cations of Takeuchi repair mainly include baffle leak and supravalvular Takeuchi procedure and concomitant mitral valve repair in our hos- . pulmonary stenosis, and the baffle leak is the most common compli- . 1,4 pital. During the operation, we found that the left coronary artery . cation (27%). We reviewed related articles in recent 20 years and (LCA) arises from the left sinus of pulmonary trunk, and we created . summarized some information about the baffle leak after Takeuchi an aortopulmonary window above the left aortic sinus and an intra- . repair of ALCAPA (see Table 1). It shows that the incidence rate of pulmonary tunnel that baffles the aorta to the ostium of the anomal- . baffle leak is about 27.8%, which is similar to the study by Ginde ous LCA through the anterior wall of the pulmonary trunk. After the et al., and 9.7% of patients received surgical reoperation. operation, she was followed regularly for 3 months and unaware of In this case, the patient was confirmed to have baffle leak 4 any abnormalities. Four months after the Takeuchi repair, she began months after the Takeuchi repair, and the diameter of the fistula to complain of chest pain on exertion. Physical examination revealed was about 4 mm, which was likely to explain the reason why the a Grade 3/6 continuous murmur of puffing character at left sternal patient felt chest pain on exertion. In addition to the patient’s Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/2/1/4956396 by Ed 'DeepDyve' Gillespie user on 03 July 2018 Transcatheter closure for baffle leak 3 Figure 1 Before the Takeuchi repair, computed tomography angiography shows that the left coronary artery arises from the main pulmonary artery. Figure 2 Four months after the Takeuchi repair, computed tomography angiography confirms a fistulous connection from the baffle into the main pulmonary artery, the diameter of the fistula is about 4 mm (A, red arrow); after the interventional therapy, the red arrow shows the device (B). symptom, this complication may result in certain sequelae if un- 54 mmHg to 68 mmHg, which means improved coronary perfusion treated, such as chronic myocardial ischaemia and angina, myocar- after the baffle leak closure, and PAP decreased due to shunt clos- dial infarction, heart failure, pulmonary hypertension, endocarditis, ure and probably improved cardiac function. To our knowledge, and so on, therefore the patient had a strong will to be treated this is the first reported case of transcatheter closure for baffle after the baffle leak was confirmed. Because of the short time after leak after Takeuchi repair. There are still no available relevant art- the first thoracotomy (only 4 months) and the difficulty, severe icles at home and abroad we can learn from. Our initial experience trauma and high risks of second thoracotomy, such as post-opera- is that: (i) make clear the relationship between the fistula and the tive adhesion, bleeding, respiratory failure, pulmonary infection, ostium of coronary, evaluate the feasibility of percutaneous trans- pulmonary atelectasis, pleural effusion, and so on, we finally catheter closure, (ii) interventional procedure should be very soft reached an agreement with cardiac surgeons and decided to treat to avoid the injury of coronary, (iii) avoid the occlusion of coronary the baffle leak through percutaneous transcatheter closure, which . after the implantation of occluder. But there are still some prob- has the advantages of lesser trauma and haemorrhage, slight pain, . lems have to be addressed, for example, what kind of baffle leak and rapid recovery. The patient was informed and willing to be . can be treated by percutaneous transcatheter closure? What type treated by this method. After the interventional therapy, the pa- . of occluder can be used for baffle leak? What size of occluder tient’s symptom improved significantly, diastolic BP increased from . should be recommended? Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/2/1/4956396 by Ed 'DeepDyve' Gillespie user on 03 July 2018 4 H. Hai-Bo et al. Figure 3 Percutaneous transcatheter closure: ascending aortic angiography in the left anterior oblique view (A) and selective coronary angiography of the left main coronary artery (B) demonstrates contrast leakage from the left coronary artery into the main pulmonary artery (red arrow); select- ive coronary angiography of the left main coronary artery in the left anterior oblique view (C) demonstrates no contrast leakage after implantation of the device (red arrow). associated text has been obtained from the patient in line with COPE Table 1 Summary of studies on baffle leak after guidance. Takeuchi repair of anomalous origin of the left coronary . artery from the pulmonary artery Conflict of interest: none declared. Study Year No. of Baffle Reoperation References patients leak . ................................................................................................. . 1. Ginde S, Earing MG, Bartz PJ, Cava JR, Tweddell JS. Late complications after 4 . Takeuchi repair of anomalous left coronary artery from the pulmonary artery: Schwartz et al. 1997 21 11 3 . case series and review of literature. Pediatr Cardiol 2012;33:1115–1123. 3 . Cochrane et al. 1998 12 2 0 . 2. Pe~ na E, Nguyen ET, Merchant N, Dennie C. ALCAPA syndrome: not just a pedi- 10 . Birk et al. 2000 7 1 1 . atric disease. RadioGraphics 2009;29:553–565. 3. Cochrane AD, Coleman DM, Davis AM, Brizard CP, Wolfe R, Karl TR. Excellent Michielon et al. 2003 12 1 1 long-term functional outcome after an operation for anomalous left coronary ar- Ojala et al. 2009 4 1 0 . tery from the pulmonary artery. J Thorac Cardiovasc Surg 1999;117:332–342. Ginde et al. 2012 9 3 1 4. Schwartz ML, Jonas RA, Colan SD. Anomalous origin of left coronary artery from pulmonary artery: recovery of left ventricular function after dual coronary Quanlin and Jie 2013 7 1 1 repair. J Am Coll Cardiol 1997;30:547–553. Total (%) 72 20 (27.8) 7 (9.7) . 5. Michielon G, Di Carlo D, Brancaccio G, Guccione P, Mazzera E, Toscano A, Di . Donato RM. Anomalous coronary artery origin from the pulmonary artery: cor- relation between surgical timing and left ventricular function recovery. Ann Thorac Surg 2003;76:581–588. . 6. Ojala T, Salminen J, Happonen J-M, Pihkala J, Jokinen E, Sairanen H. Excellent functional result in children after correction of anomalous origin of left coronary Conclusion artery from the pulmonary artery—a population-based complete follow-up study. Interact Cardiovasc Thorac Surg 2010;10:70–75. Percutaneous transcatheter closure is an alternative treatment option . 7. Yu-Juan Q, Pei-Jun L. Advancement of surgical therapy for anomalous left coron- for the baffle leak after Takeuchi repair of ALCAPA, this case is just an . ary artery from pulmonary artery. Chin J Clin Thorac Cardiovasc Surg 2017;24: 239–242. initial exploration, more experience, and clinical data are still needed. . . 8. Takeuchi S, Imamura H, Katsumoto K, Hayashi I, Katohgi T, Yozu R, Ohkura M, Inoue T. New surgical method for repair of anomalous left coronary artery from pulmonary artery. J Thorac Cardiovasc Surg 1979;78:7–11. 9. Dua R, Smith JA, Wilkinson JL, Menahem S, Karl TR, Goh TH, Mee RBB. Long- Supplementary material . term follow-up after two coronary repair of anomalous left coronary artery from the pulmonary artery. J Cardiac Surg 1993;8:384–390. Supplementary material is available at European Heart Journal – Case 10. Birk E, Stamler A, Katz J, Berant M, Dagan O, Matitiau A, Erez E, Blieden LC, . Vidne BA. Anomalous origin of the left coronary artery from the pulmonary ar- Reports online. tery: diagnosis and postoperative follow up. Isr Med Assoc J 2000;2:111–114. 11. Quanlin M, Jie X. Surgical treatment of anomalous origin of the left coronary Consent: The author/s confirm that written consent for submis- artery from the pulmonary artery. Chin J Ethnomed Ethnopharm 2013;15: . 42–43. sion and publication of this case report including image(s) and Downloaded from https://academic.oup.com/ehjcr/article-abstract/2/2/1/4956396 by Ed 'DeepDyve' Gillespie user on 03 July 2018

Journal

European Heart Journal - Case ReportsOxford University Press

Published: Mar 29, 2018

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