Spontaneous closure of a coronary artery fistula in childhood

Summary An infant with a symptomatic coronary artery fistula, documented by angiography, is presented. By age 5 years, clinical evidence of the fistula was no longer present. Repeat cardiac catheterization confirmed spontaneous closure of the coronary artery fistula. More information regarding the natural history of a coronary artery fistula is required before an elective surgical approach can be recommended in all asymptomatic children. Supported in part by Research Grants from the National Institute of Health. Training Grant HL 07413-03     

小儿先天性动静脉瘘的诊治

目的 探讨对小儿先天性动静脉瘘(congenital arteriovenous fistula，CAVF)的诊断与治疗。方法 本组共收治15例CAVF患儿，男6例、女9例，年龄8～14岁。对全部患儿行多普勒超声，动脉造影或MRA检查。发现病变局限者5例，病变范围广泛性者10例，病变大多发生于四肢。动脉造影显示：动静脉之间出现瘘支；动脉分支多而紊乱，可呈碗蜒扭曲状，病变多为弥漫性；静脉象早期出现。对11例患儿行手术治疗，其中4例局限性病变者行病灶切除。对7例病变广泛者行1或多次减量手术，方法是沿动脉主干切除瘘支与病变组织，对分流量大又无法切除的病变组织予以缝扎，以减少分流量。对5例患儿行栓塞治疗(其中2例为术前栓塞治疗以减少手术出血)。1例未治疗。结果 随访1～6年。5例局限性患儿中3例治愈，2例病情控制。10例广泛性病变中，5例病情好转或控制，5例病情仍继续发展。结论 影像学检查是CAVF诊断与治疗的依据。CAVF治疗困难。尤其对广泛性病变者，无论手术或栓塞治疗疗效均欠理想。但对小儿CAVF应尽早治疗，这样可控制病情发展．防止并发症发生。     

Failure of color flow mapping to identify coronary artery to pulmonary artery fistula

Summary A case of coronary artery to pulmonary artery fistula presented with the typical clinical findings of asymptomatic precordial continuous murmur and small left-to-right shunt. Echocardiography with color flow mapping failed to demonstrate the fistula on three occasions, including one study performed after angiographic demonstration of the fistula site. This case illustrates the importance of angiography in patients with unexplained continuous murmurs, even when echocardiographic and color flow mapping results are normal.     

Successful percutaneous closure of a coronary artery fistula with a detachable balloon

Precutaneous transcatheter closure technique of a coronary artery fistula with a detachable balloon was performed for a 14 year old male student. Complete closure of the fistula without any complications was confirmed by angiography after the procedure. When the patient underwent a second angiography 6 months after the closure, it was confirmed that the position of the balloon had not changed, that the interruption of the flow of the fistula had been maintained and that the diameter of the left coronary artery and the fistula were reduced. Percutaneous closure technique using a detachable balloon may become the primary treatment for a coronary artery fistula in place of surgical ligation.     

Coronary artery fistula complicating the evaluation of Kawasaki disease

Summary Two patients clinically diagnosed with Kawasaki disease were found to have a coronary artery to pulmonary artery fistula. The dilemma of deciding the etiology of coronary artery dilatation in these patients is discussed along with management.     

Traumatic bilateral vertebral artery dissection at the dural entry point site in a 10-year-old boy

We present a 10-year-old boy who was admitted with headache and neurological symptoms after a trauma in the schoolyard. Cerebral MRI revealed an extensive ischaemia in the bilateral cerebellar hemispheres, left middle cerebellar peduncle, and right vermis. Digital substraction angiography demonstrated bilateral vertebral artery dissections at the dural entry point site. This case emphasises the management of patients with traumatic vertebral artery dissection.     

经导管介入法治疗小儿先天性冠状动脉瘘9例

目的探讨经导管介入法治疗小儿先天性冠状动脉瘘的方法及效果。方法广东省人民医院2002年1月至2005年1月收治的先天性冠状动脉瘘患儿9例,先行心导管检查及选择性冠状动脉造影,显示瘘管的形态及引流情况,建立达到或通过瘘管欲堵闭处的输送轨道。6例瘘管最窄处内径≤3mm的病例选用可控弹簧圈进行堵闭,3例内径>3mm的病例应用Amplazter动脉导管堵闭器进行堵闭。结果8例堵闭成功,1例瘘管扭曲严重的病例堵闭失败而行外科手术。全部病例随诊2个月至3年,随诊中无并发症发生。结论经导管介入治疗冠状动脉瘘是一种创伤性小、疗效确切的方法,但必须严格掌握介入治疗适应证。     

Transcatheter embolization of coronary artery fistula with controlled release coils

ABSTRACT The treatment for large congenital coronary cameral fistulas has been surgical but with advances in interventional catheterization techniques transcatheter embolization of these fistulas with coils or detachable balloons is now possible. This report describes occlusion of a congenital coronary arteriovenous fistula in a 6-year-old girl.     

Arteriovenous malformations as a cause of congestive heart failure in the newborn and infant

Three cases of congestive heart failure are presented caused by a cerebral arteriovenous aneurysm, a pulmonary arteriovenous aneurysm and a placental angioma respectively. Such conditions should be borne in mind in otherwise unexplained heart failure in the neonatal period and in infants. The haemodynamic influence of the malformation depends on its anatomy, giving rise to various clinical pictures.     

Pulmonary atresia and ventricular septal defect with coronary artery to pulmonary artery fistula: Case report and review of the literature

The pulmonary blood supply in patients with pulmonary atresia and ventricular septal defect is highly variable. Several cases have been reported in the literature in which a coronary artery to pulmonary artery fistula, alone or in combination with additional vessels, supplies the pulmonary circulation. We report a case of successful repair during early infancy, with unique hemodynamic, clinical, and anatomic features. The literature is reviewed.     

Coil embolization of a congenital intrahepatic arterioportal fistula: increasing experience in management

Congenital intrahepatic arterioportal fistula (IAPF) is a rare condition and there is limited experience of transcatheter embolization. We report here the transcatheter coil embolization of an incidentally found, asymptomatic congenital IAPF in a 16-month-old patient. After demonstrating that the IAPF was fed by two branches of a dilated left hepatic artery and drained into an aneurysm of the left portal vein, the feeding arteries were superselectively catheterized and occluded by microcoils in a single session. Follow-up was uneventful for 1 year. With respect to this and previously reported cases, we believe that embolization of feeding arteries is a safe and effective method that should be the primary choice of treatment; however, further cases should be reported to confirm this belief.     

Catheter embolization of pulmonary arteriovenous fistulas in an infant

Summary An infant with pulmonary arteriovenous fistulas is described. Surgical ligation of the fistulas and limited resection of paraenchyma from the right lung was followed at 11 months of age, by successful steel coil embolization of residual fistulas.     

Surgical therapy of a coronary artery fistula draining into the left atrium

Coronary artery fistulas (CAFs) are rare vascular anomalies. We describe a case of CAF draining into the left atrium, combined with an atrial septum defect (ASD) and azygos continuity in a 3-year-old girl. Surgical therapy included closure of the ASD combined with closure of the CAF through a left atrial approach. Even though CAF can be treated by interventional means, surgical correction may be required in the presence of associated cardiac abnormalities.     

Giant cutaneous haemangioma with heart failure in a neonate: successful surgical treatment

A large cutaneous haemangioma was identified on the posterior thorax of an 18-weeks' gestation fetus during routine antenatal ultrasonography. The diagnosis was confirmed after birth with computed axial tomography. Intractable heart failure was cured by excision of the haemangioma. Offprint requests to: E. R. Howard     

不同类型先天性冠状动脉瘘介入治疗随访研究

目的 评价儿童不同类型冠状动脉瘘(CAF)介入封堵治疗的近中期疗效、并发症及抗凝治疗方案。方法 回顾性分析2006 年1 月至2014 年1 月行CAF 介入封堵治疗的12 例患儿术前、造影及术后随访资料,记录不同类型CAF 的封堵方式、抗凝方案、术后并发症、辅助检查结果。结果 本组患儿年龄1~158 个月;近端型/中型4 例,近端型/大型5 例,远端型/中型3 例,均成功封堵;术后随访3.5±2.4 年;11 例患儿术后口服阿司匹林6 个月,1 例口服18 个月;无血栓、介入并发症,术后左心室射血分数、心胸比、肺动脉压下降,病变冠状动脉开口直径减小。结论 儿童期介入封堵治疗近端型和远端型/中型CAF 近中期疗效、安全性满意;术后阿司匹林抗凝治疗可预防近中期血栓事件,但疗程和安全性有待进一步随访研究。     

A case of right coronary artery to left ventricle fistula: Two-dimensional echocardiographic study

Summary A 12-year-old boy with a decrescendo diastolic murmur maximal at the lower right sternal border was suspected of having a coronary arteriovenous fistula. Cardiac catheterization and angiography revealed a right coronary artery to left ventricle fistula with marked dilatation of the right coronary artery.This is the thirteenth case of this anomaly reported in the literature. Characteristic two-dimensional echocardiographic features of this case are presented. A markedly dilated right coronary artery was revealed by two-dimensional echocardiography to take a normal right coronary artery course and empty into the posteromedial portion of the left ventricle.This case indicates the possibility of visualizing a dilated peripheral portion of the right coronary artery using two-dimensional echocardiography.     

Vertebral and carotid artery anomalies in patients with aberrant right subclavian arteries

Background There is little published evidence regarding the patterns and prevalence of vertebral artery (VA) and common carotid artery (CCA) anomalies in patients with an aberrant right subclavian artery (ARSCA). Objective To study the patterns and prevalence of VA and CCA anomalies in patients with ARSCA. Materials and methods In a 2-year period we reviewed the children referred with suspected vascular ring who had undergone multidetector-row CT. Patients with ARSCA were reviewed for VA and CCA patterns and their prevalence and relevance were calculated. Results In total, 102 patients with ARSCA were identified. VA anomalies were present in 16 patients (15.7%), and CCA anomalies (common carotid trunk) in 21 patients (20.6%). In some patients with VA anomalies, the right VA arose from the right CCA and in some the left VA arose from the aortic arch. When the left VA arose from the aortic arch it was situated between the left CCA and the left SCA or between the left SCA and the ARSCA. Conclusion If neurointerventionalists understand these potential anomalies and their prevalence, time and contrast medium could be saved when catheterizing the VA and CCA in patients with ARSCA.