J Cardiol 2007 Jul; 50（1）: 71 – 76

Cardiac Tamponade Due toRupture of Coronary Artery
Fistulas With a Giant Aneurysm
Containing a Free Floating Ball
Thrombus : A Case ReportYuji

IWASAWA, MD

YutakaKITAMURA, MD

Kikuhiko

HIGUMA, MD

Fumiaki

ONO, MD

Kiyotaka

IMOTO, MD＊1

Kazuo

KIMURA, MD, FJCC＊2

Abstract ─────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────────
A 76-year-old woman was admitted to our hospital with cardiac tamponade. Chest computed tomography revealed a large tumor adjacent to the pulmonary artery, the center of which was demonstrated as ahigh density area without contrast medium. Coronary angiography revealed a large aneurysm consisting ofcoronary artery fistulas. An afferent vessel connected tangentially to the surface of the aneurysm, so blood
flowed around along the inner wall of the aneurysm. A filling defect was demonstrated in the center of theaneurysm that indicated a thrombus. The cause of cardiac tamponade was seemed to be bleeding of thecoronary artery fistulas. Coil embolization was performed, but recanalization occurred. Therefore, ligatureof the fistulas and aneurismal resection were performed using a cardiopulmonary bypass. A fresh floating
thrombus was found in the aneurysm.
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Key Words■ Aneurysms■ Cardiac tamponade■ Thrombosis（floating thrombus）■ Coronary vessels（coronary artery fistula）

INTRODUCTION
Coronary artery fistula is a rare anomaly thatconsists of a communication between the coronary
arteries and a cardiac chamber or great vessel, andis usually asymptomatic. The clinical presentations,which are mainly dependent on the severity of theleft-to-right shunt, include fatigue, dyspnea, andangina. Potential complications include pulmonary
hypertension, congestive heart failure, myocardialischemia due to coronary steal phenomenon, infec-tive endocarditis, and rupture or thrombosis of thefistulas.1）
We report a case of rupture of coronary artery
fistulas with a giant aneurysm containing a free
floating ball thrombus manifesting as cardiac tamponade.
CASE REPORT
A 76-year-old woman with a history of hypertension was admitted to our hospital with dizzinessand hypotension. On admission, her blood pressure──────────────────────────────────────────────
横須賀市立市民病院 循環器科 : 〒 240−0195 神奈川県横須賀市長坂 1−3−2 ; 横浜市立大学附属市民総合医療センター心臓血管センター ＊ 1 心臓血管外科，＊ 2 循環器内科，横浜Department of Cardiology, Yokosuka Municipal Hospital, Kanagawa ; Departments of ＊ 1Cardiovascular Surgery and ＊ 2Cardiology,
Cardiovasular Center, Yokohama City University Medical Center, Yokohama
Address for correspondence : IWASAWA Y, MD, Department of Cardiology, Yokosuka Municipal Hospital, Nagasaka 1−3−2,
Yokosuka, Kanagawa 240−0195 ; E-mail : yiwasawa-circ@umin.ac.jp
Manuscript received February 28, 2007 ; revised April 7, 2007 ; accepted April 9, 200771

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Iwasawa, Kitamura, Higuma et al

Fig. 1 Doppler echocardiogram（left）and schematic representation（right）
from the parasternal shortaxis view
Echo free space and a large tumor in front of the basal left ventricle were seen. A 2 cm mass was detected in
the tumor.
LV ＝ left ventricle ; RV ＝ right ventricle ; PE ＝ pericardial effusion ; T ＝ tumor ; M ＝ mass.

Fig. 2 Chest computed tomography scans
Without contrast medium（left）, early phase（middle）and late phase（right）with contrast medium, showing
the large tumor adjacent to the pulmonary artery.

was 92/68 mmHg and pulse rate was 106 beats/min.
Her physical examination was remarkable for acontinuous murmur of Levine Ⅱ at the left upper
sternal border and jugular venous distention.
Paradoxical pulse was not apparent. Echocardiography revealed massive pericardial effusionand collapse of the right ventricle during early diastole. A large（5 × 5 cm in diameter）tumor wasdetected in the front of the basal left ventricle.
Abnormal blood flow signals and a 2 cm mass weredetected in the tumor（Fig. 1）. Chest computed
tomography（Fig. 2）revealed the tumor adjacent tothe pulmonary artery. The center of the tumor wasdemonstrated as a high density area without contrast medium. The tumor was enhanced by contrast
medium like the great vessels except for the center
J Cardiol 2007 Jul; 50（1）: 71 – 76

Rupture of Coronary Artery Fistulasof the tumor. The computed tomography number ofthe pericardial effusion was 48.6, indicating hemopericardium. Chest T1-weighted magnetic resonance imaging（ Fig. 3）showed the left anterior
descending coronary arteries below the tumor, but
could not detect the continuity between the tumor
and the coronary arteries.
We considered that the cause of the cardiac tamponade was bleeding of the tumor. Urgent coronary
angiography established the differential diagnosis
from malignancy. Left coronary angiography（Fig. 4）showed a large aneurysm arising from theproximal part of the left anterior descending arteryand draining into the pulmonary artery. An afferent
vessel ran straight and connected with the bottomof the aneurysm tangentially, so contrast medium
flowed along the inner wall of the aneurysm. A
round filling defect was demonstrated in the centerof the aneurysm. Smaller fistulas, which had notformed aneurysms, were also detected between theright coronary artery and the pulmonary artery. AFig. 3 Chest T1-weighted magnetic resonanceimage
The tumor is located in front of the basal left
ventricle with pericardial effusion.

Fig. 4 Left coronary angiograms
A large aneurysm arose from the proximal part of the left anterior descending artery. An afferent vessel
was straight and connected with the bottom of the aneurysm tangentially, and contrast medium showedcounterclockwise flow along the inner wall of the aneurysm（A, B, C, D in order）. A round filling defectwas seen in the center of the aneurysm（E）, schema of blood flow in the aneurysm（F）.

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73

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Iwasawa, Kitamura, Higuma et alA

BCDFig. 5 Operative photographs（A, C, D）and schematic representation（B）
A : A large（5 cm in diameter）aneurysm of coronary artery fistula was located between the pulmonary
artery and left anterior descending coronary artery.
B : RV ＝ right ventricle ; Ao ＝ ascending aorta ; PA ＝ pulmonary artery ; LAD ＝ left anterior descending
coronary artery.
C : Soft thrombus was detected in the aneurysm.
D : Aneurysm inner wall.

significant rise in oxygen saturation was observedin the pulmonary artery, and the left-to-right shuntratio was 19%. The clinical diagnosis was cardiac
tamponade due to rupture of the coronary artery fistulas with a giant aneurysm containing a floatingball thrombus.
We performed coil embolization of the fistulas
and drainage of 300 ml bloody pericardial effusion,
when her blood pressure was normalized（134/60 mmHg）. Two weeks after the treatment,
coronary angiography revealed recanalization of
the embolization site. We performed ligature of thefistulas originating from the right and left coronary
arteries, and resection of the aneurysm using cardiopulmonary bypass（ Fig. 5）. The resected
aneurysm showed smooth arterial wall and no atherosclerotic change such as calcification.

Histological study of the aneurysm wall revealed
trilaminar structure and inflammatory cellular infiltration in the adventitia. The rupture site could not
be detected. A fresh free floating ball thrombus（Fig. 6）
was found in the aneurysm. The postoperative course was uneventful, and she was discharged
home.
DISCUSSION
Coronary artery fistulas are identified incidentally in 0.3−0.8% of routine cardiac angiographic
series.1）The right coronary artery is the site of thefistula in about 55% of cases, the left coronary
artery in about 35%, and both coronary arteries in5%. 1）Aneurysmal formation is present in 19−26%,2,3）and rupture of the fistulas, pericardial effusion, and sudden death are the presenting feaJ Cardiol 2007 Jul; 50（1）: 71 – 76

Rupture of Coronary Artery Fistulas

Fig. 6 Photograph of the free-floating ball thrombus in
the aneurysmtures.4,5）Thrombosis of the fistulas is also recognized. However, this case involved an unusual free
floating ball thrombus in the aneurysm of the coronary artery fistulas. In this case, an afferent vessel
connected tangentially to the surface of aneurysm,
so blood flowed around along the inner wall of theaneurysm. The free floating ball thrombus wasformed in the center of the aneurysm where theblood flow stagnated. We were misled by the presence of this free floating ball thrombus. The thrombus was very fresh, so appeared as high density on
computed tomography without contrast medium
and the aneurysm was well enhanced, mimicking amalignant tumor in which bleeding occurred.
Coronary angiography was most valuable for thediagnosis in this case. Cardiac catheterization may
be indispensable for the diagnosis and hemodynamic evaluation of coronary artery fistulas. Various
cardiac imaging modalities are utilized for adjunctdiagnostic evaluation. Two-dimensional echocar要

diography can usually detect an enlarged feeding
vessel with an abnormal flow pattern. 6,7）
Multiplanar transesophageal echocardiography also
accurately defines the origin, course, and drainagesite of the fistulas.8）Ultrafast computed tomography or magnetic resonance imaging with contrast
medium can evaluate the course of abnormal tortuous blood vessels using multiplanar reconstruction.9,10）However, several limitations1）have been
described with these imaging techniques. Various
modalities should be used to establish the diagonsis.
Most adult patients with coronary artery fistulas
remain free from symptoms for long periods. 11）
Small fistulas may close spontaneously, especiallyin pediatric patients. 12） The management forasymptomatic patients is still controversial. Patientswith complications need to undergo surgical or percutaneous intervention. Surgical closure13）and percutaneous transcatheter closure14）are both safe andeffective. Transcatheter closure has been proposedas the initial choice, even if staged procedure suchas surgical intervention is ultimately required.15）In
our patient, recanalization was recognized after coilembolization, possibly because the afferent vesselof the aneurysm was straight and tapering, andblood flow velocity in the afferent vessel was relatively high. If recanalization occurs after transcatheter closure, we should not hesitate to performthe surgical management of a patient with severe
complications like this case.
The present case of cardiac tamponade due torupture of coronary artery fistula with a giant
aneurysm containing a giant thrombus floatedshows that early diagnosis and early treatment isimportant to manage this very rare, but severe complication.

約巨大瘤を伴う冠動脈瘻の破裂により心タンポナーデをきたした 1 例:
瘤内球状浮遊血栓の合併
岩澤祐二

北 村豊

小野文明

井元清隆

日隈菊比児
木村一雄

症例は心タンポナーデを呈した 76 歳，女性．胸部コンピューター断層撮影（CT）で血性心餒液貯留と肺動脈に接する径 5 cm の腫瘤が認められた．腫瘤中心部は単純像で高濃度を呈し，腫瘤内部

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76

Iwasawa, Kitamura, Higuma et alの辺縁寄りは濃く造影された．冠動脈造影検査の結果，この腫瘤は冠動脈瘻の巨大瘤と診断され，心タンポナーデの原因は瘻破裂による出血と考えられた．瘤への流入血管は瘤壁表面を直線的に走行し接線方向に開口しており，瘤内の血流が内壁に沿って円周状に渦巻くように観察された．血流の滞る中心部に島状の陰影欠損が認められ，単純 CT で高濃度を呈したものは浮遊血栓と思われた．
コイル塞栓術後に心餒ドレナージが行われたが，塞栓部位の再疎通が認められ，人工心肺下に冠動脈瘻結紮術と瘤切除術が施行された．瘤内から比較的新鮮な球状浮遊血栓が摘出された．冠動脈瘻の破裂はまれであり，さらに本例は冠動脈瘻の巨大瘤と瘤内の特異な血流パターンのために形成された球状浮遊血栓を観察しえた貴重な症例であった．

J Cardiol 2007 Jul; 50（1）: 71−76

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