両側冠動脈肺動脈瘻に肝動静脈瘻を伴った1例

A case of bilateral coronary artery to pulmonary artery fistulas associated with hepatic arteriovenous fistula

　先天性冠動脈瘻は，先天性心疾患の中では比較的まれな疾患であるが，選択的冠動脈造影法の普及により数多く報告され，臨床的にも注目されるようになった。その頻度は，先天性心疾患の0.2ないし0.5％といわれ^1,2），左右両側からの冠動脈瘻はさらにその1ないし5％と少ない^3,4）。今回我々は，両側冠動脈肺動脈瘻に肝動静脈瘻を合併した1例を経験した。両者の合併した例は，これまで文献上報告がみられず，まれな症例と思われるので報告する。

A 55-year-old man, previously treated for prima-ry hypothyroidism, was admitted for evaluation of atypical chest pain. On physical examination, the pulse rate was 60 and blood pressure was 132/ 84 mmHg. Heart sounds were normal and no mur-mur was heard. Abdominal palpation showed no abnormal finding, and bruit was not heard. The electrocardiogram was normal and stress-induced is-chemia was not found. The chest X-ray showed nosign of pulmonary hyperaemia or mass lesion, and central shadow was normal (CTR=44%). During right heart catheterization, the pressures were nor-mal in the cardiac chambers, pulmonary artery, and wedge position. A significant increase in oxygen saturation was disclosed at pulmonary artery level (10%) and the upper site of the inferior vena cava (22%). Coronary arteriography found coronary artery fistulas from the left main trunk, the left anterior descending artery, the left circumflex, and the right coronary artery to the pulmonary trunk.The left and right ventriculograms were normal. The digital subtraction angiography of celiac artery showed hepatic arteriovenous fistula. Ultrasonogra-phy and computed tomography found no mass lesion of the liver. Since combination of the bilateral coronary artery with pulmonary artery fistula, and hepatic arteriovenous fistula is very rare, the pre-sent case is worth noting for the investigation of the pathogenesis of congenital arteriovenous fistu-las.