儿童先天性冠状动脉瘘46例介入治疗及中短期随访

doi:10.12372/jcp.2022.21e1291

摘要/Abstract

摘要：

目的总结儿童先天性冠状动脉瘘（CAF）的介入治疗效果、并发症及随访情况。方法回顾性分析2013年1月至2021年1月收治的先天性CAF患儿的临床资料。结果纳入46例CAF患儿，男22例、女24例，中位年龄36.0（24.5~49.5）月，中位体重14.9（12.2~20.0）kg。瘘管引流口直径大小为3.4（3.0~4.4）mm；受累冠脉有不同程度增宽，开口处内径6.1（4.5~7.8）mm。46例CAF患儿中，9例行冠脉造影后评估为介入封堵困难，最终37例施行介入封堵术，其中35例成功封堵。建立动静脉轨道（A-V LOOP）逆向封堵者15例，经主动脉端正向封堵者20例；使用动脉导管未闭封堵器8例，室间隔缺损封堵器2例，弹簧圈3例，血管塞22例。35例封堵成功患儿术后出现即刻残余分流10例，8例随访中微小残余分流消失，2例末次随访时仍有残余分流。术后血栓形成2例。结论介入封堵是治疗儿童先天性CAF安全有效的手段，但需充分评估CAF的走行、开口以及侧枝血管情况，以提高手术成功率；对于存在血栓形成风险的患儿，可能需延长术后常规抗凝疗程。

关键词: 冠状动脉瘘, 介入治疗, 残余分流, 血栓, 儿童

Abstract:

Objective To analyze the efficacy,complications and prognosis of transcatheter closure (TCC) of congenital coronary artery fistula (CAF) in children. Methods Clinical data of patients diagnosed with CAF and underwent TCC from January 2013 to January 2021 were retrospectively analyzed. Results A total of 46 patients with CAF were enrolled in the study, including 22 boys and 24 girls, with median age of 36.0 (24.5-49.5) months and median body weight of 14.9 (12.2-20.0) kg. In 46 patients with CAF, the fistula shunt was3.4 (3.0-4.4) mm, and there were varying degrees of widening of the involved coronary artery with an internal diameter of 6.1 (4.5-7.8) mm at the opening. It was considered difficult to perform TCC in the other 9 patients. Among the 35 patients who underwent successful TCC, arteriovenous loop was established to perform retrograde closure in 15 patients. The other 20 patients underwent antegrade closure through the aorta. PDA device (8 cases), VSD device (2 cases), coil (3 cases) and vascular plug (22 cases) were used in these patients.Among the 35patients who underwent successful TCC, 10 patients showed immediate residual shunt after closure. Tiny residual shunt disappeared during follow-up in eight patients. Two patients still showed residual shunt at the last follow-up. Thrombosis formation was found in two cases. Conclusion TCC is a safe and effective method for the treatment of CAF in children. It is necessary to fully evaluate the course, opening and collateral vessels of CAF to improve the success of intervention. For patients suffering risk of thrombosis, a longer course of routine anticoagulation after TCC may be required.

Key words: coronary artery fistula, transcatheter closure, residual shunt, thrombosis formation, child

刘月, 朱荻绮, 李奋, 傅立军, 刘廷亮, 郭颖, 高伟, 黄美蓉, 沈捷. 儿童先天性冠状动脉瘘46例介入治疗及中短期随访[J]. 临床儿科杂志, 2022, 40(11): 843-847.

LIU Yue, ZHU Diqi, LI Fen, FU Lijun, LIU Tingliang, GUO Ying, GAO Wei, HUANG Meirong, SHEN Jie. Transcatheter closure and short- and medium-term follow-up of 46 children with congenital coronary artery fistula[J]. Journal of Clinical Pediatrics, 2022, 40(11): 843-847.

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参考文献 23

[1]	Collins N, Mehta R, Benson L, et al. Percutaneous coronary artery fistula closure in adults: technical and procedural aspects[J]. Catheter Cardiovasc Interv, 2007, 69(6): 872‐880. doi: 10.1002/ccd.21085
[2]	Ilkay E, Celebi OO, Kacmaz F, et al. Percutaneous closure of coronary artery fistula: long-term follow-up results[J]. Postepy Kardiol Interwencyjnej, 2015, 11(4): 318‐322.
[3]	Wang C, Zhou K, Li Y, et al. Percutaneous transcatheter closure of congenital coronary artery fistulae with patent ductus arteriosus occluder in children: focus on patient selection and intermediate-term follow-up results[J]. J Invasive Cardiol, 2014, 26(7): 339‐346. pmid: 24993992
[4]	Gowda ST, Latson LA, Kutty S, et al. Intermediate to long-term outcome following congenital coronary artery fistulae closure with focus on thrombus formation[J]. Am J Cardiol, 2011, 107(2): 302‐308. doi: 10.1016/j.amjcard.2010.09.018 pmid: 21211608
[5]	Budnur SC, Singh B, Jayaranganath M. Percutaneous transcatheter device closure of coronary arteriovenous fistula through persistent left superior vena cava[J]. J Invasive Cardiol, 2013, 25(2): E24‐E26. pmid: 23388233
[6]	Ghaffari S, Akbarzadeh F, Pourafkari L. Aneurysmal coronary arteriovenous fistula closing with covered stent deployment: a case report and review of literature[J]. Cardiol J, 2011, 18(5): 556‐559. pmid: 21947993
[7]	Lee ML, Chen M. Diagnosis and management of congenital coronary arteriovenous fistula in the pediatric patients presenting congestive heart failure and myocardial ischemia[J]. Yonsei Med J, 2009, 50(1): 95‐104. doi: 10.3349/ymj.2009.50.1.95
[8]	Buccheri D, Chirco PR, Geraci S, et al. Coronary Artery Fistulae: Anatomy, Diagnosis and Management Strategies[J]. Heart Lung Circ, 2018, 27(8): 940-951. doi: S1443-9506(18)30029-5 pmid: 29503240
[9]	Warnes CA, Williams RG, Bashore TM, et al. ACC/AHA 2008 guidelines for the management of adults with congenital heart disease: a report of the American College of Cardiology/American Heart Association Task Force on Practice Guidelines (Writing Committee to Develop Guidelines on the Management of Adults With Congenital Heart Disease). Developed in Collaboration With the American Society of Echocardiography, Heart Rhythm Society, International Society for Adult Congenital Heart Disease, Society for Cardiovascular Angiography and Interventions, and Society of Thoracic Surgeons[J]. J Am Coll Cardiol, 2008, 52(23): e143-e263. doi: 10.1016/j.jacc.2008.10.001
[10]	Armsby LR, Keane JF, Sherwood MC, et al. Management of coronary artery fistulae. Patient selection and results of transcatheter closure[J]. J Am Coll Cardiol, 2002, 39(6): 1026‐1032. pmid: 11897446
[11]	Qureshi SA, Tynan M. Catheter closure of coronary artery fistulas[J]. J Interv Cardiol, 2001, 14(3): 299‐307. pmid: 12053388
[12]	Liu X, Zhang L, Qi Z, et al. The characteristics of coronary-pulmonary artery fistulas and the effectivity of trans-catheter closure: a single center experience[J]. J Thorac Dis, 2019, 11(7): 2808‐2815. doi: 10.21037/jtd.2019.06.60 pmid: 31463109
[13]	Mavroudis C, Backer CL, Rocchini AP, et al. Coronary artery fistulas in infants and children: a surgical review and discussion of coil embolization[J]. Ann Thorac Surg, 1997, 63(5): 1235‐1242. pmid: 9146308
[14]	Latson LA. Coronary artery fistulas: how to manage them[J]. Catheter Cardiovasc Interv, 2007, 70(1): 110‐116.
[15]	Zhu XY, Zhang DZ, Han XM, et al. Transcatheter closure of congenital coronary artery fistulae: immediate and long-term follow-up results[J]. Clin Cardiol, 2009, 32(9): 506‐512. doi: 10.1002/clc.20650
[16]	Jama A, Barsoum M, Bjarnason H, et al. Percutaneous closure of congenital coronary artery fistulae: results and angiographic follow-up[J]. JACC Cardiovasc Interv, 2011, 4(7): 814‐821. doi: 10.1016/j.jcin.2011.03.014
[17]	Xiao Y, Gowda ST, Chen Z, et al. Transcatheter closure of coronary artery fistulae: considerations and approaches based on fistula origin[J]. J Interv Cardiol, 2015, 28(4):380-389. doi: 10.1111/joic.12212 pmid: 26086715
[18]	Collins N, Benson LN, Horlick EM. Iatrogenic ST elevation during percutaneous closure of a coronary artery fistula[J]. Congenit Heart Dis, 2012, 7(1): 80‐83. doi: 10.1111/j.1747-0803.2011.00527.x pmid: 21702886
[19]	Kharouf R, Cao QL, Hijazi ZM. Transcatheter closure of coronary artery fistula complicated by myocardial infarction[J]. J Invasive Cardiol, 2007, 19(5): E146‐E149. pmid: 17470976
[20]	Ascoop AK, Budts W. Percutaneous closure of a congenital coronary artery fistula complicated by an acute myocardial infarction[J]. Acta Cardiol, 2004, 59(1): 67‐69. doi: 10.2143/AC.59.1.2005161
[21]	Thakkar B, Patel N, Poptani V, et al. Clinical and angiographic follow-up of coronary artery fistula interventions in children: techniques and classification revisited[J]. Cardiol Young, 2015, 25(4): 670‐680. doi: 10.1017/S1047951114000614 pmid: 24775405
[22]	El-Sabawi B, Al-Hijji MA, Eleid MF, et al. Transcatheter closure of coronary artery fistula: a 21-year experience[J]. Catheter Cardiovasc Interv, 2020, 96(2): 311-319. doi: 10.1002/ccd.28721
[23]	Newburger JW. Kawasaki disease: medical therapies[J]. Congenit Heart Dis, 2017, 12(5): 641-643. doi: 10.1111/chd.12502 pmid: 28580631

项目	倒数(%)
起源血管
左冠状动脉	22(47.8)
左回旋支	14(30.4)
左前降支	4(8.7)
左主干	3(6.5)
左回旋支+左前降支	1(2.2)
右冠状动脉	24(52.2)
引流部位
右心房	16(34.8)
右心室	28(60.8)
左心房	1(2.2)
左心室	1(2.2)