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Ann Thorac Surg 2000;70:1394-1397
© 2000 The Society of Thoracic Surgeons

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Case report

Successful surgical treatment of giant coronary artery aneurysm with fistula

^a Department of Thoracic and Cardiovascular Surgery, Sapporo Medical University School of Medicine, Sapporo, Japan

Address reprint requests to Dr Mawatari, Department of Thoracic and Cardiovascular Surgery, Sapporo Medical University School of Medicine, South 1 West 16, Chuo-ku, Sapporo, 060-8556 Japan
e-mail: mawatari{at}sapmed.ac.jp

	Abstract

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Giant coronary artery aneurysm with fistula formation is a rare entity. We report a giant coronary artery aneurysm with a maximum diameter of 70 mm with fistula, in which a favorable course was obtained after surgical treatment. We also review the literature on giant coronary artery aneurysms exceeding 50 mm in maximum diameter.

	Introduction

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Coronary artery aneurysm (CAA) is a disease that most cardiologists encounter when the the size of the aneurysm is small. Rarely the aneurysm is giant, has a fistula, and requires operation. In this report, we describe such a case.

The patient was a 56-year-old woman who was found to have an abnormal chest roentgenogram obtained at the time of a medical examination. Coronary angiography and echocardiography demonstrated an anomaly of the right coronary artery (RCA). The patient was transferred to our hospital for further evaluation. She had no subjective symptoms and no abnormal hematologic findings. Physical examination revealed a continuous grade II/VI murmur at the left fourth intercostal space. Her medical and familial histories were unremarkable. Chest roentgenography showed a projection to the right of the cardiac shadow. Chest computed tomographic (CT) scan and three-dimensional CT demonstrated an abnormal shadow dorsad to the right atrium (RA) and caudad to the right pulmonary artery (Fig 1). Echocardiography revealed dilatation of the RCA ostium. Coronary angiography (Fig 2) showed that the RCA was dilated beginning at the ostium and had a serpiginous appearance. The dilated portion formed a giant CAA and supplied an RCA of normal size. The aneurysm had a fistula opening into the RA. Oxygen saturation was increased in the RA (inflow of RA: 80.4%, outflow of RA: 92.8%). The left coronary artery appeared normal. On the basis of these findings, a diagnosis of right giant CAA with fistula into the RA was made, and operation was advised.

View larger version (59K): [in this window] [in a new window]   Fig 1. (A) Computed tomograph (CT) showing the coronary artery aneurysm. (B) Three-dimensional CT shows the coronary artery aneurysm that exists at dorsad to the right atrium and caudad to the right pulmonary artery. (Ao = aorta; CAA = coronary artery aneurysm; RA = right atrium; RPA = right pulmonary artery.)

View larger version (140K): [in this window] [in a new window]   Fig 2. Angiogram of the right coronary artery shows the giant coronary artery aneurysm (black arrowheads) with a dilated serpiginous appearance (white arrowheads).

The postoperative course was uneventful. Angiography and echocardiography revealed no abnormal communication related to the aneurysm and the dilated portion of the RCA; the bypass graft was patent.

	Comment

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The incidence of CAA is reported between 0.3% to 4.9% for patients undergoing coronary angiography [1, 2]. On the other hand, congenital coronary artery fistula is found in only 0.2% of patients who have coronary angiography [3]. Therefore, giant CAA with fistula formation is rare. The main causes of CAA include atherosclerosis, congenital Kawasaki disease, postpercutaneous transluminal coronary angioplasty, and endocarditis [4]. Most of the previously reported cases of CAA with fistula are congenital. The present case is also most probably congenital because of the patient’s history, the form of the lesion, and the pathological findings.

With regard to size, a diameter of more than 8 mm in infants is considered as "giant CAA" [5]. In adults, the reported diameter of giant CAA varies. Therefore we listed cases of "giant CAA" from MEDLINE and selected English articles describing CAAs with diameters exceeding 50 mm. These cases are presented in Table 1, (total 17, including this one). There is no sex or age predilection. Six cases were subclinical, and 11 were associated with symptoms such as chest discomfort, chest pain, and palpitation. These symptoms may be caused by coronary ischemia, or heart failure. Twelve aneurysms originated from the RCA and five from the left coronary artery. Cases in which the aneurysm originated from the proximal part of the RCA were predominant.

Five cases were associated with a fistula; four of these were congenital, and the other, was atherosclerotic in origin. In the congenital cases, the coronary artery was dilated from the ostium.

Complications of CAA are rupture, angina, and so on, and in cases with fistula formation, congestive heart failure and infectious endocarditis. With regard to therapy, simple observation may be justifiable for small aneurysms that produce no symptoms. However, an operation should be considered for large aneurysms that produce marked symptoms. The procedure for dealing with coronary aneurysm includes resection or plication of the aneurysm and isolation of blood flow to the aneurysm. In cases with fistula, closure of the fistula is needed. When native coronary blood flow is compromised, a bypass graft is essential. Management of the cases in Table 1 approximately followed these lines.

The present case was asymptomatic, but we opted to operate because of the large size of the aneurysm and high risk of rupture. The left-right shunt ratio was 20%; thus there was a risk of congestive heart failure. Furthermore, the aneurysm compromised normal blood flow to the RCA.

	References

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1. Oliveros R.A., Falsetti H.L., Carroll R.J., Heinle R.A., Ryan G.F. Atherosclerotic coronary artery aneurysm. Arch Intern Med 1974;134:1072-1076.[Abstract/Free Full Text]
2. Swaye P.S., Fisher L.D., Litwin P., et al. Aneurysmal coronary artery disease. Circulation 1983;67:134-138.[Abstract/Free Full Text]
3. Wenger NK. Rare causes of coronary artery disease. In: Hurst JW, ed. The heart. New York: McGraw-Hill, 1978:1348–9.
4. Vranckx P., Pirot L., Benit E. Giant left main coronary artery aneurysm in association with severe atherosclerotic coronary disease. Cathet Cardiovasc Diagn 1997;42:54-57.[Medline]
5. Gersony W.M. Diagnosis and management of Kawasaki disease. JAMA 1991;265:2699-2703.[Abstract/Free Full Text]

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Kiyofumi Morishita Tomio Abe Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Mawatari, T. Articles by Abe, T. Search for Related Content PubMed PubMed Citation Articles by Mawatari, T. Articles by Abe, T.