CORONARY arteriovenous fistulas are uncommon, but their detection has. Rupture of a Giant Saccular Aneurysm of Coronary Arteriovenous Fistulas

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1 Rupture of a Giant Saccular Aneurysm of Coronary Arteriovenous Fistulas Masahiro ITO, MD, Makoto KODAMA, MD, Makihiko SAEKI, 1 MD, Hiroshi FUKUNAGA, MD, Tomoji GOTO, 2 MD, Hidenori INOUE, 2 MD, Shigetaka KASUYA, 2 MD, and Yoshifusa AIZAWA, MD SUMMARY A 58-year-old Japanese woman was admitted to our hospital because of chest pain. A continuous murmur was detected at the left parasternal area. Electrocardiogram showed ST elevation in leads V 2, V 3 and V 4. Chest computed tomography and echocardiography demonstrated pericardial effusion and a large mass which was adjacent to the pulmonary artery. An abnormal blood flow was detected in the mass by Doppler echocardiography. Coronary angiography confirmed that the mass was a giant aneurysm of coronary arteriovenous fistula arising from both the left and right coronary arteries. This patient had no symptoms until rupture of the fistula. Rupture of a coronary arteriovenous fistula is very rare but can be a cause of chest pain and pericardial effusion. (Jpn Heart J 2000; 41: ) Key words: Coronary arteriovenous fistula, Aneurysm, Rupture, Chest pain, Pericardial effusion CORONARY arteriovenous fistulas are uncommon, but their detection has increased because of the widespread use of coronary angiography. 1) Therapeutic strategies for coronary arteriovenous fistulas have not been clearly defined because the natural histories of the disorder are still uncertain. 2) The majority of coronary arteriovenous fistulas are small and asymptomatic, especially in young patients. In older patients, both symptoms and fistula-related complications increase. 3) Surgical repair for coronary arteriovenous fistulas is generally recommended in the presence of symptoms of heart failure or large left-to-right shunts. 4) On the other hand, the management for asymptomatic patients with a coronary arteriovenous fistula is controversial. We report the rupture of a giant aneurysm of a coronary arteriovenous fistula presented by chest pain with pericardial effusion. The patient had no fistula-related symptoms until the rupture. From the First Department of Internal Medicine, Niigata University School of Medicine, 1 Department of Internal Medicine, Nagaoka Central General Hospital, 2 Department of Cardiovascular Surgery, Tachikawa General Hospital, Niigata, Japan. Address for correspondence: Masahiro Ito, MD, First Department of Internal Medicine, Niigata University School of Medicine, Asahimachi, Niigata, Niigata , Japan. Received for publication March 16, Revised and accepted June 14,

2 660 ITO, ET AL Jpn Heart J September 2000 CASE REPORT A 58-year-old woman was referred from another hospital because of chest pain with pericardial effusion. She had a history of hypertension for 10 years and chronic renal failure on hemodialysis for 2 years. She had no history of chest pain. Her blood pressure on admission was 106 / 76 mmhg. A continuous murmur of Levine 2 / 6 was detected in the left parasternal area. The chest X-ray showed an abnormal shadow adjacent to the left pulmonary artery which was detected 3 years prior to the present episode (Figure 1A, B). Laboratory data showed anemia (red blood cells 292 / mm 3, hemoglobin 9.5 g / dl, hematocrit 29.6%), as well as increases in the serum level of creatinine (5.5 mg / dl) and blood urea nitrogen (28.5 mg / dl). Creatine kinase was within the normal range (84 IU / l). The electrocardiogram (ECG) recorded at another hospital showed sinus rhythm with a heart rate of 50 beats / min, and ST elevation was present in leads V 2, V 3 and V 4 but disappeared by the time of admission to our hospital. Chest computed tomography and echocardiography demonstrated pericardial effusion and a large mass with a maximum diameter of 4.0 cm adjacent to the pulmonary artery (Figure 2). Blood flow signals were also detected in the mass by Doppler echocardiography. Emergent cardiac catheterization was performed and a significant rise in oxygen saturation was observed in the pulmonary artery; the pulmonary-to-systemic flow ratio was 1.4. Coronary angiography confirmed that the mass was Figure 1. A: Chest X ray at admission showed an abnormal shadow (white arrow). B: Chest X ray taken three years before this event also showed an abnormal shadow (white arrow).

3 Vol 41 No 5 RUPTURE OF CORONARY ARTERIOVENOUS FISTULA 661 Figure 2. Chest computed tomography at admission showed pericardial effusion and a mass shadow. A large mass was adjacent to the pulmonary artery (white arrow). Figure 3. Coronary angiography showed that the mass was a giant aneurysm of a coronary arteriovenous fistula arising from the left coronary artery. A minor fistula was detected from the right coronary artery. a giant saccular aneurysm of a coronary arteriovenous fistula arising from the left anterior descending artery draining into the pulmonary artery (Figure 3). A smaller fistula was also detected between the right coronary artery and the pulmonary artery. No leakage of contrast media into the pericardial space could be verified. Since the patient's status was stable and pericardial effusion gradually decreased after admission, elective sur-

4 662 ITO, ET AL Jpn Heart J September 2000 Figure 4. Echocardiography before failing blood pressure (A) and after failing blood pressure (B) on day 6. Echocardiography showed an increase in pericardial effusion. gery was planned. On day 6, the patient's systolic blood pressure suddenly fell to 80 mmhg and she experienced chest pain. Echocardiography showed an increase in the echo-free space (Figure 4). Diastolic compression of both the right atria and ventricle was not clear. The patient was diagnosed as having a re-rupture of a coronary fistula, and underwent surgery on Day 7. A pin-hole rupture was detected during surgery on the giant aneurysm of the coronary arteriovenous fistulas arising from the left anterior descending artery. Both fistulas arising from the left and right coronary arteries were ligated and the aneurysm resected. The operative and postoperative course were uneventful and she was discharged. DISCUSSION Coronary arteriovenous fistulas have become widely recognized because of the widespread use of coronary angiography. 1) The symptoms of coronary arteriovenous fistulas are variable and depend on the patient's age. 3) Young patients are generally asymptomatic. More than half of the older patients have symptoms such as dyspnea, fatigue and anginal chest pain. There are many complications associated with coronary arteriovenous fistulas, including congestive heart failure, angina pectoris, myocardial infarction, endocarditis, pulmonary hypertension, and rupture. 4) These complications increase with the age of the patients. 3,5) Early diagnosis of the existence of a coronary arteriovenous fistula is important. A continuous murmur and abnormal chest X-ray shadow may suggest the

5 Vol 41 No 5 RUPTURE OF CORONARY ARTERIOVENOUS FISTULA 663 presence of coronary arteriovenous fistulas. 2,6) This case had both findings. The most difficult problem concerning management of a patient with a coronary arteriovenous fistula is the surgical indication. Surgery is justified when the patient has a large left-to-right shunt or symptoms of heart failure. 4) Management for asymptomatic patients has remained controversial because there are various natural histories as mentioned above. The rupture of a coronary arteriovenous fistula, including an aneurysmal fistula, is a very rare complication but might be fatal. 7,8) An aneurysmal formation was present in 26% of patients with a coronary arteriovenous fistula. 2) Some cases have reported that an aneurysm had progressively enlarged during their follow-up period. 9,10) In this case, chest X rays suggest that the saccular aneurysm has significantly enlarged for only 3 years. There are no defined risk factors in predicting the rupture of an aneurysm of a coronary arteriovenous fistula. Lowe, et al. recommended that all patients with major coronary fistulas should be considered for surgical correction, and that the closure should be electively performed before symptoms and pathologic changes in the heart become apparent. 1) However, it has been reported that small fistulas have a relatively benign natural history, and thus require little surgical treatment. 11) Surgical closure of a coronary arteriovenous fistula has been reported to be safe and effective, although there are occasional complications. 1,3,5,12) In this case, the patient had neither symptoms nor complications related to coronary arteriovenous fistulas until the rupture. It is difficult to recommend surgical treatment for all asymptomatic patients with a coronary arteriovenous fistula. Close observation is important, especially if there is an aneurysm. We think that early surgical treatment must be recommended if a saccular aneurysm shows progressive dilatation during the follow-up period. In summary, we have reported rupture of a giant aneurysm of coronary arteriovenous fistulas. The rupture of coronary arteriovenous fistulas is very rare but can be a cause of chest pain and pericardial effusion. Early diagnosis of the existence of a coronary arteriovenous fistula is important. REFERENCES 1. Lowe JE, Oldham HN, Jr, Sabiston DC, Jr. Surgical management of congenital coronary artery fistulas. Ann Surg 1981; 194: Said SAM, El Gamal MIH. Coronary angiographic morphology of congenital coronary arteriovenous fistulas in adults. Cathet Cardiovasc Diagn 1995; 35:

6 664 ITO, ET AL Jpn Heart J September Liberthson RR, Sagar K, Berkoben JP, Weintraub RM, Levine FH. Congenital coronary arteriovenous fistula. Circulation 1979; 59: Friedman WF. Diseases of the heart, pericardium, aorta, and pulmonary vascular bed. In: Braunwald E, editor. Heart Disease, 5th edition. Philadelphia: W.B. Saunders Company 1997; Oldham HN Jr, Ebert PA, Young WG, Sabiston DC Jr. Surgical management of congenital coronary artery fistula. Ann Thorac Surg 1971; 12: Engle MA, Goldsmith EI, Holswade GR, Goldberg HP, Glenn F. Congenital coronary arteriovenous fistula. Diagnostic evaluation and surgical correction. N Engl J Med 1961; 264: Habermann JH, Howard ML, Johnson ES. Rupture of the coronary sinus with hemopericardium. Circulation 1963; 28: Bauer HH, Allmendinger PD, Flaherty J, Owlia D, Rossi MA, Chen C. Congenital coronary arteriovenous fistula: Spontaneous rupture and cardiac tamponade. Ann Thorac Surg 1996; 62: Okita Y, Miki S, Kusuhara K, et al. Aneurysm of coronary arteriovenous fistula presenting as a calcified mediastinal mass. Ann Thorac Surg 1992; 54: Tayama E, Ohashi M, Fukunaga S, et al. Surgical treatment of a coronary artery fistula with concomitant saccular coronary artery aneurysm. Jpn Circ J 1999; 63: Gillebert C, Van Hoof R, Van de Werf F, Piessens J, De Geest H. Coronary artery fistulas in an adult population. Eur Heart J 1986; 7: Urrutia-S CO, Falaschi G, Ott DA, Cooley DA. Surgical management of 56 patients with congenital coronary artery fistulas. Ann Thorac Surg 1983; 35: