Ann Thorac Surg 2007;83:1532-1534
© 2007 The Society of Thoracic Surgeons
Case Reports
Hybrid Therapy for Rapid Enlargement of Hibernating Coronary Arteriovenous Fistulas
Shizu Oyamada, MDa,
Junjiro Kobayashi, MDa,
Osamu Tagusari, MDa,*,
Kazuo Niwaya, MDa,
Hiroyuki Nakajima, MDa,
Shunichi Miyazaki, MDb,
Kohji Kimura, MDc,
Shigeyuki Echigo, MDd,
Soichiro Kitamura, MDa
a Department of Cardiovascular Surgery, National Cardiovascular Center, Osaka, Japan
b Department of Internal Medicine, National Cardiovascular Center, Osaka, Japan
c Department of Radiology, National Cardiovascular Center, Osaka, Japan
d Department of Pediatrics, National Cardiovascular Center, Osaka, Japan
Accepted for publication October 2, 2006.
* Address correspondence to Dr Tagusari, National Cardiovascular Center, Cardiovascular Surgery, 5-7-1 Fujishirodai, Suita, Osaka 565-8565 Japan (Email: otagusar{at}hsp.ncvc.go.jp).
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Abstract
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The use of hybrid therapy for recurrent multiple coronary arteriovenous fistulas in a 56-year-old woman is reported. The patient underwent surgical closure of a coronary arteriovenous fistula of the right coronary artery under cardiopulmonary bypass at 47 years of age. Reoperation was required 9 years later for recurrence of the same fistula. It was divided under a beating heart. Early postoperative angiography showed complete occlusion of the right coronary fistula. However, hibernating fistulas of the left circumflex artery, which had been left untouched because of insignificant shunt with no remarkable change for 9 years, increased in size rapidly. Transcatheter embolization was successfully performed for these residual fistulas.
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Introduction
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Surgery for coronary arteriovenous fistula (CAVF) can be accomplished with very low morbidity and mortality [1–4]. Complete surgical closure of multiple CAVFs is sometimes difficult, however, because of complex anatomy and often, multiple sites of origin. This report describes a case of successful hybrid therapy in a 56-year-old patient with a late recurrent fistula of the right coronary artery (RCA) and hibernating fistulas of the left circumflex artery that showed rapid enlargement soon after closure of the RCA fistula.
A 46-year-old woman was referred to our institution for evaluation of a murmur. Selective coronary angiography visualized a large RCA fistula draining into the coronary sinus (Fig 1A). Small fistulas at distal sites of the circumflex artery were also noticed (Fig 1B). Oximetry demonstrated a 2.5:1 left-to right shunt.
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Fig 1. Coronary angiography before the initial operation. (A) A large right coronary fistula drained into the coronary sinus. (B) Fistulas of the left circumflex coronary artery were small.
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At the age of 47 years, the patient underwent surgical correction under cardiopulmonary bypass and cardioplegia. The dilated RCA near the crux was opened, and the proximal opening of the fistula was closed directly. The circumflex fistulas were left untouched because of insignificant shunt. The patient made an uneventful recovery. Postoperative catheterization showed only a 1.08:1 left-to-right shunt despite a small residual shunt of the RCA.
When the patient was 56 years old, she repeatedly underwent cardiac catheterization because follow-up echocardiograms showed increased shunting. Coronary angiography showed similar findings to those in previous examinations of fistulas from both the RCA (Fig 2A) and circumflex artery (Fig 2B). Oximetry revealed a 1.72:1 left-to-right shunt.
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Fig 2. Coronary angiography before the second operation. (A) A recurrent right coronary fistula showed large shunting. (B) Circumflex fistulas showed no remarkable change and were still insignificant.
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Reoperation was performed through a repeat median sternotomy. The dilated RCA was shown to pursue a tortuous course along the epicardial surface of the right atrioventricular groove. Extracardiac dissection and isolation of the fistula were performed using a Starfish (Medtronic, Minneapolis, MN) heart positioner. The thickened fistula was divided after temporary occlusion of the fistula showed no ischemic response. Meanwhile, the circumflex fistulas were left because the shunt was insignificant, with no remarkable change for 9 years after the first preoperative coronary angiography.
On postoperative day 9, angiography showed that the dilated proximal RCA was occluded by thrombus (Fig 3A), and the distal RCA was filled by collateral arteries from the circumflex artery. Oximetry results almost normalized, with a 1.1:1 left-to-right shunt. An unexpected finding was that the sizes of the circumflex fistulas were significantly increased (Fig 3B). According to the coronary angiography findings, the fistulas had three feeding arteries from the end of the circumflex artery draining into the coronary sinus.
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Fig 3. Coronary angiography after the second operation. (A) The dilated right coronary artery was occluded by thrombus. (B) Hibernating fistulas rapidly increased in size after closure of the main fistula.
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A percutaneous transcatheter embolization was therefore performed to occlude all feeding arteries, which was accomplished with 19 microcoils (GDCTM Fibered VortX Shape, Boston Scientific, Natick, MA; Fig 4). Two years after hybrid therapy, the patient’s condition was good, and follow-up echocardiogram showed no significant shunting.
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Fig 4. Left coronary angiography after coil embolization. Circumflex fistulas were occluded by percutaneous transcatheter embolization.
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Comment
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Coronary arteriovenous fistula is a rare congenital anomaly that can be complicated by congestive heart failure, angina, endocarditis, or rupture of a coronary aneurysm. Hence, early surgical intervention has been recommended to prevent the development of significant and potentially fatal complications. In addition, recent reports have emphasized the efficacy of percutaneous transcatheter techniques for the treatment of CAVF. The clinical decision of intervention or surgery is usually based on angiographic findings or an abnormal hemodynamic status.
In this patient, there were two issues in the clinical course. The first was the recurrence of the RCA fistula after the first operation. In the angiographic findings before the second operation, the location of the shunt flow was similar to that of the angiographic findings before the first operation. On the basis of these results, the cause of recurrence might have been dehiscence of friable tissue that was directly closed. The second issue was the rapid enlargement of the hibernating circumflex fistulas after the redo operation.
It is generally accepted that small asymptomatic fistulas should be managed conservatively because of the possibility of spontaneous closure [5, 6]; therefore, the circumflex fistulas, which were small and showed no remarkable change for 9 years, were ignored at the second operation. Regarding the potential long-term risk of enlargement, we considered elective transcatheter embolization. However, early postoperative angiography revealed that the hibernating fistulas rapidly increased in size, while the main fistula in the opposite coronary system disappeared and the dilated proximal RCA was thrombosed. Moreover, the distal RCA was opacified owing to retrograde flow from collateral arteries from the circumflex fistulas, which had not been recognized before. This showed that collateral circulation from the circumflex coronary system had rapidly increased to maintain the blood supply of the thrombosed RCA.
The phenomena of proximal thrombosis and distal circulation filled by collaterals have been reported as prevalent findings [4], but in this case we report opposite coronary fistulas hibernating for 9 years that showed rapid enlargement soon after closure of the main fistula. The precise mechanism of this rapid enlargement is difficult to prove, but we propose that the possible mechanism is the acute change of turbulent flow in the coronary sinus by abrupt termination of coronary blood flow from the RCA.
Transcatheter embolization was finally adopted to treat these multiple, growing fistulas. Several criteria exist for the performance of coil occlusion, such as absence of a single narrow drainage site, absence of large branch vessels, and safe access to the coronary artery. Close follow-up is also required because the long-term outcome after coil embolization remains unknown [7, 8]. Hybrid therapy should, however, be considered the treatment of choice for coronary arteriovenous fistulas that are difficult to close completely by surgical intervention because of complex anatomy and multiple sites of origin.
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References
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Abstract
Introduction
