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法洛四联症合并肺动脉瓣缺如婴儿的反复肺出血:经弹簧圈封堵体肺侧支动脉的介入治疗

Recurrent pulmonary haemorrhage in an infant with tetralogy of Fallot and absent pulmonary valve: interventional treatment by coil occlusion of systemic-to-pulmonary collateral arteries.

作者信息

Kumpf Matthias, Schäfer Jürgen, Hofbeck Michael

机构信息

Department of Pediatric Cardiology, Pulmology and Intensive Care, Tuebingen, Germany.

出版信息

Cardiol Young. 2013 Jun;23(3):443-6. doi: 10.1017/S1047951112000935. Epub 2012 Jul 17.

Abstract

Although clinically silent in the majority of cases, enlarged bronchial arteries or systemic-to-pulmonary collateral arteries may complicate congenital heart disease in infants, causing significant left-to-right shunting with subsequent pulmonary congestion and respiratory compromise. So far, pulmonary haemorrhage, a well-known complication in older patients with cyanotic congenital heart disease, has not been described in infancy. We describe the case of a 6-month-old girl with tetralogy of Fallot and absent pulmonary valve who developed haemoptysis with severe respiratory distress following corrective surgery of the cardiac malformation. High-resolution computed tomography of the thorax followed by selective angiography revealed a systemic-to-pulmonary collateral artery originating from the left internal mammary artery. Pulmonary haemorrhage stopped immediately following coil occlusion of the collateral. A second episode of pulmonary haemorrhage occurred at the age of 9 months during mechanical ventilation for treatment of pneumonia. Repeat angiography revealed two more collateral vessels. Again coil occlusion resulted in prompt resolution of pulmonary haemorrhage. According to our experience, enlarged bronchial arteries or systemic-to-pulmonary collateral arteries should be considered in infants with cyanotic heart disease with unexplained pulmonary congestion or prolonged respiratory problems.

摘要

尽管在大多数情况下临床上并无症状,但支气管动脉增粗或体肺侧支动脉可能会使婴儿先天性心脏病病情复杂化,导致大量左向右分流,继而引起肺充血和呼吸功能受损。到目前为止,肺出血作为大龄青紫型先天性心脏病患者一种众所周知的并发症,在婴儿期尚未见报道。我们报告一例6个月大患有法洛四联症且肺动脉瓣缺如的女孩,在心脏畸形矫正手术后出现咯血并伴有严重呼吸窘迫。胸部高分辨率计算机断层扫描及随后的选择性血管造影显示,一条体肺侧支动脉起源于左乳内动脉。在用弹簧圈封堵该侧支动脉后,肺出血立即停止。在9个月大因肺炎接受机械通气治疗期间发生了第二次肺出血。再次血管造影显示又有两条侧支血管。再次用弹簧圈封堵后,肺出血迅速得到缓解。根据我们的经验,对于患有青紫型心脏病且伴有无法解释的肺充血或长期呼吸问题的婴儿,应考虑存在支气管动脉增粗或体肺侧支动脉的情况。

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