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Myocardial ischemia assessed by dobutamine stress echocardiography in a patient with bicoronary to pulmonary artery fistulas
Myocardial Ischemia Assessed by Dobutamine Stress Echocardiography in a Patient with Bicoronary to Pulmonary Artery Fistulas Abdou Elhendy, MD, PhD, Peter R Nierop, MD, Jos R.T.C. Roelandt, MD, PhD, and Paolo M Fioretti, MD, PhD, Rotterdam, the Netherlands
A congenital coronary artery fistula is a rare anomaly characterized by a communication between one or more coronary arteries and a cardiac chamber, coronary vein, or less frequently, the pulmonary artery. The reported complications o f this anomaly are congestive heart failure, infective endocarditis, and myocardial infarction. 2Mthough angina is not an infrequent complaint in the adult population with coronary to pulmo-
nary artery fistulas, 4 objective evidence o f myocardial ischemia in the absence of concomitant atherosclerotic coronary artery disease has not been described. In this report, we describe an adult patient with chest pain and bicoronary to pulmonary artery fistulas in whom myocardial ischemia was documented by high-dose dobutamine stress echocardiography. (J Am Soc Echocardiogr 1997;10:189-91.)
CASE REPORT
sion scintigraphy revealed no significant abnormality. Because of the submaximal heart rate attained with exercise, the patient underwent high-dose dobutamine (up to 40 /xg/kg/minute) - atropine (0.5 mg) stress echocardiography. Her heart rate increased from 66 to 142 beats/ minute. The blood pressure at rest was 124/78 mm H g and 105/49 mm H g at peak stress. No significant arrhythmia occurred during the test. The results of the resting echocardiogram were normal. During the stress echocardiography, we observed a gradual reduction of motion and thickening of the inferior wall and the basal part of the posterior septum progressing to dyskinesis (Figure 2). The test was considered to indicate the presence of myocardial ischemia in the area of the right coronary artery. This diagnosis was made by two independent observers who were unaware of the clinical, angiographic, or scintigraphic data of the patient, and who agreed on the presence and localization of abnormalities. The patient continued her medical treatment, which consisted of beta blockers, and experienced partial symptomatic relie£
A 48-year-old woman presented with exertional chest pain that was progressing to occur even at rest. She was admitted to the intensive care unit with the diagnosis of unstable angina. The results of a physical examination were unremarkable apart from a systolic murmur grade 2 / 6 in the second left intercostal space. The electrocardiogram obtained during an episode of her chest pain revealed T wave inversion in the precordial leads reverting to the upright position after the episodes of pain. The levels of cardiac enzymes were normal. Coronary angiography showed no significant narrowing of the coronary arteries. Numerous small fistulous communications between the proximal left anterior descending (LAD) and pulmonary arteries were detected. A single small fistula also was detected communicating between the left circumflex coronary artery and the pulmonary artery. Right coronary angiography revealed a large fistula originating from the ostium of the right coronary artery and communicating with the pulmonary artery (Figure 1). To assess the functional significance of anginal chest pain and coronary abnormalities, the patient underwent a bicycle exercise99mTechnetium MIBI single photon- emission computed tomography (SPECT) study after stabilization of symptoms with medical treatment. The heart rate of the patient increased from 65 to 120 beats/ minute and the test was terminated because of fatigue without precordial symptoms. The blood pressure rose from 110/70 to 180/110 mm H g at peak exercise. PerfuFrom the Thoraxcenter, University Hospital Rotterdam-Dijkzigt and Erasmus University, Rotterdam, the Netherlands. Reprint requests: Paolo M Fioretti, MD, PhD, Istituto di Cardiologia, Azienda Ospedaliera S. Maria della Miseficordia, P. le S. Maria della Misericordia 15, 33100 Udine, Italy. Copyright © 1997 by the American Society of Echocardiography. 0894-7317/97 $5.00 + 0 27/4/75613
DISCUSSION T h e correlation between s y m p t o m s o f angina and specific markers o f myocardial ischemia is important to clarify the functional significance o f angina in adult patients with coronary artery fistulas) ,5 So far, myocardial ischcmia has n o t been d o c u m e n t e d in patients with coronary to p u l m o n a r y artery fistulas in the absence o f c o n c o m i t a n t atherosclerotic disease o f the coronary artery. Few reports have described the presence o f objective evidence o f myocardial ischcmia in the m o r e c o m m o n type o f fistulas c o m m u n i c a t i n g with cardiac chambers or coronary veins. Brooks and 189
190
Elhendy et al.
Journal of the AmericanSocietyof Echocardiography March 1997
F i g u r e 1 Right coronary artery injection showing a large fistula originating from the right coronary ostium (arrow).
F i g u r e 2 Two-dimensional echocardiographic images at rest and during high-dose dobutamine stress from the apical four-chamber view (Top) and apical two-chamber view (Bottom), showing dyskinesis of the basal posterior septum and basal inferior wall at high-dose dobutamine stress (arrows).
Journal of the AmericanSocietyof Echocardiography Volume10 Number2
Bates 6 described two patients with congenital coronary artery fistulas communicating with the left ventricle without concomitant athcrosclerotic narrowing. These patients had significant electrocardiographic changes during ischemic episodes. Glynn ct al. 7 reported on a patient with a fistula communicating between the LAD artery and coronary vein. This patient had a reversible perfusion defect that was detected on exercise thallium scintigraphy. In contrast, Said et al. reported that exercise M I B I scintigraphy failed to reveal myocardial ischemia in a 49year-old w o m a n with atypical chest pain and multiple coronary-pulmonary fistulas. 8 In our study patient, myocardial ischemia was docu m e n t e d by the presence o f transient wall motion abnormalities in the right coronary artery territory during dobutamine stress echocardiography. The occurrence o f myocardial ischemia confined to the right coronary artery territory could be explained by the smaller size o f the fistulas o f the LAD and the left circumflex arteries. The lower pealc exercise rate compared with the peak dobutamine heart rate could not explain the conclusive findings o f myocardial ischemia on dobutamine stress echocardiography as opposed to exercise myocardial perfusion scintigraphy. A stronger, inotropic effect of dobutamine compared with exercise could explain the discrepancy between the outcome o f the dobutamine and exercise stress test, because the double product at peak stress was lower during the dobutamine test. Myocardial ischemia in patients with coronary fistulas is t h o u g h t to be caused by coronary steal. 6,7 Therefore, transient mechanical dysfunction mostly implies inadequacy o f myocardial supply relative to oxygen demand during stress and represents a highly specific marker for myocardial ischemia. 9 For that purpose, in patients with limited exercise capacity, dobutamine stress testing may be a first choice test.9,10
CONCLUSION
We d o c u m e n t e d myocardial ischemia in an adult patient who presented with angina and was found to
Eihendy et al.
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have bicoronary to pulmonary artery fistulas. The occurrence o f transient wall motion abnormalities during high-dose dobutamine stress echocardiography in this patient showed that flow malperfusion may result in true ischemia during stress. Assessment o f myocardial ischemia with dobutamine stress echocardiography may be helpful in the evaluation o f anginal symptoms in patients with coronary artery fistulas. We are grateful to Jan Tuin and Ren~ Frowyn for the preparation of the figures.
REFERENCES
1. Libcrthson RR, SagarK, Berkoben JP, Weintraub ItaM,Levine FH. Congenital coronary arteriovenous fist-ulac. Circulation 1979;59:849-54. 2. Yamanaka O, Hobbs RE. Coronary artery abnormalities in 126,595 patients undergoing coronary angiography. Cathet Cardiovasc Diagn 1990;21:28-40. 3. AshrafSS,Shaukat N, Fisher M, ClarkeB, KeenanDJ. Bicoronary-pulmonaryfistulae with coexistent mitral valveprolapse: a case report and literature review of coronary-pulmonary fistula. Eur Heart I 1994;15:571-4. 4. Urmita-S CO, Fallaschi G, Ott DA, Cooley DE. Surgical management of 56 patients with congenital coronary artery fistulas. Ann Thorac Surg t983;35:300-7. 5. Said SA, Landman GH. Coronary-pulmonary fistula: Long term follow-up in operated and in non-operated patients. Int J Cardiol 1990;27:203-i0. 6. Brooks CH, Bates PD. Coronary artery-leftventricular fistula with angina pectoris. Am Heart J 1983;I06:404-6. 7. Glynn TP, Fleming RG, Haist ~L, Hunteman RK. Coronary arteriovenous fistula as a cause for reversible thallium-201 perfusion defect. J Nucl Med 1994;35:i808-10. 8. Said SA, Bucx JJ, van de Well FA. Stress MIBI scintigraphyin multiple coronary-pulmonaryfistula: failure to demonstrate 'steal' phenomenon. Int J Cardiol 1992;35:270-2. 9. SalustriA, Fioretti PM, Pozzoli MMA, McNeill AJ, Roelandt JRTC. Dobutamine stress echocardiography: its role in the diagnosis of coronary artery disease. Eur Heart I 1992;13: 70-7. 10. RuffoloRR. The pharmacologyofdobutamine. Am 5r Med Sci 1987;294:244-8.
A congenital coronary artery fistula is a rare anomaly characterized by a communication between one or more coronary arteries and a cardiac chamber, coronary vein, or less frequently, the pulmonary artery. The reported complications o f this anomaly are congestive heart failure, infective endocarditis, and myocardial infarction. 2Mthough angina is not an infrequent complaint in the adult population with coronary to pulmo-
nary artery fistulas, 4 objective evidence o f myocardial ischemia in the absence of concomitant atherosclerotic coronary artery disease has not been described. In this report, we describe an adult patient with chest pain and bicoronary to pulmonary artery fistulas in whom myocardial ischemia was documented by high-dose dobutamine stress echocardiography. (J Am Soc Echocardiogr 1997;10:189-91.)
CASE REPORT
sion scintigraphy revealed no significant abnormality. Because of the submaximal heart rate attained with exercise, the patient underwent high-dose dobutamine (up to 40 /xg/kg/minute) - atropine (0.5 mg) stress echocardiography. Her heart rate increased from 66 to 142 beats/ minute. The blood pressure at rest was 124/78 mm H g and 105/49 mm H g at peak stress. No significant arrhythmia occurred during the test. The results of the resting echocardiogram were normal. During the stress echocardiography, we observed a gradual reduction of motion and thickening of the inferior wall and the basal part of the posterior septum progressing to dyskinesis (Figure 2). The test was considered to indicate the presence of myocardial ischemia in the area of the right coronary artery. This diagnosis was made by two independent observers who were unaware of the clinical, angiographic, or scintigraphic data of the patient, and who agreed on the presence and localization of abnormalities. The patient continued her medical treatment, which consisted of beta blockers, and experienced partial symptomatic relie£
A 48-year-old woman presented with exertional chest pain that was progressing to occur even at rest. She was admitted to the intensive care unit with the diagnosis of unstable angina. The results of a physical examination were unremarkable apart from a systolic murmur grade 2 / 6 in the second left intercostal space. The electrocardiogram obtained during an episode of her chest pain revealed T wave inversion in the precordial leads reverting to the upright position after the episodes of pain. The levels of cardiac enzymes were normal. Coronary angiography showed no significant narrowing of the coronary arteries. Numerous small fistulous communications between the proximal left anterior descending (LAD) and pulmonary arteries were detected. A single small fistula also was detected communicating between the left circumflex coronary artery and the pulmonary artery. Right coronary angiography revealed a large fistula originating from the ostium of the right coronary artery and communicating with the pulmonary artery (Figure 1). To assess the functional significance of anginal chest pain and coronary abnormalities, the patient underwent a bicycle exercise99mTechnetium MIBI single photon- emission computed tomography (SPECT) study after stabilization of symptoms with medical treatment. The heart rate of the patient increased from 65 to 120 beats/ minute and the test was terminated because of fatigue without precordial symptoms. The blood pressure rose from 110/70 to 180/110 mm H g at peak exercise. PerfuFrom the Thoraxcenter, University Hospital Rotterdam-Dijkzigt and Erasmus University, Rotterdam, the Netherlands. Reprint requests: Paolo M Fioretti, MD, PhD, Istituto di Cardiologia, Azienda Ospedaliera S. Maria della Miseficordia, P. le S. Maria della Misericordia 15, 33100 Udine, Italy. Copyright © 1997 by the American Society of Echocardiography. 0894-7317/97 $5.00 + 0 27/4/75613
DISCUSSION T h e correlation between s y m p t o m s o f angina and specific markers o f myocardial ischemia is important to clarify the functional significance o f angina in adult patients with coronary artery fistulas) ,5 So far, myocardial ischcmia has n o t been d o c u m e n t e d in patients with coronary to p u l m o n a r y artery fistulas in the absence o f c o n c o m i t a n t atherosclerotic disease o f the coronary artery. Few reports have described the presence o f objective evidence o f myocardial ischcmia in the m o r e c o m m o n type o f fistulas c o m m u n i c a t i n g with cardiac chambers or coronary veins. Brooks and 189
190
Elhendy et al.
Journal of the AmericanSocietyof Echocardiography March 1997
F i g u r e 1 Right coronary artery injection showing a large fistula originating from the right coronary ostium (arrow).
F i g u r e 2 Two-dimensional echocardiographic images at rest and during high-dose dobutamine stress from the apical four-chamber view (Top) and apical two-chamber view (Bottom), showing dyskinesis of the basal posterior septum and basal inferior wall at high-dose dobutamine stress (arrows).
Journal of the AmericanSocietyof Echocardiography Volume10 Number2
Bates 6 described two patients with congenital coronary artery fistulas communicating with the left ventricle without concomitant athcrosclerotic narrowing. These patients had significant electrocardiographic changes during ischemic episodes. Glynn ct al. 7 reported on a patient with a fistula communicating between the LAD artery and coronary vein. This patient had a reversible perfusion defect that was detected on exercise thallium scintigraphy. In contrast, Said et al. reported that exercise M I B I scintigraphy failed to reveal myocardial ischemia in a 49year-old w o m a n with atypical chest pain and multiple coronary-pulmonary fistulas. 8 In our study patient, myocardial ischemia was docu m e n t e d by the presence o f transient wall motion abnormalities in the right coronary artery territory during dobutamine stress echocardiography. The occurrence o f myocardial ischemia confined to the right coronary artery territory could be explained by the smaller size o f the fistulas o f the LAD and the left circumflex arteries. The lower pealc exercise rate compared with the peak dobutamine heart rate could not explain the conclusive findings o f myocardial ischemia on dobutamine stress echocardiography as opposed to exercise myocardial perfusion scintigraphy. A stronger, inotropic effect of dobutamine compared with exercise could explain the discrepancy between the outcome o f the dobutamine and exercise stress test, because the double product at peak stress was lower during the dobutamine test. Myocardial ischemia in patients with coronary fistulas is t h o u g h t to be caused by coronary steal. 6,7 Therefore, transient mechanical dysfunction mostly implies inadequacy o f myocardial supply relative to oxygen demand during stress and represents a highly specific marker for myocardial ischemia. 9 For that purpose, in patients with limited exercise capacity, dobutamine stress testing may be a first choice test.9,10
CONCLUSION
We d o c u m e n t e d myocardial ischemia in an adult patient who presented with angina and was found to
Eihendy et al.
191
have bicoronary to pulmonary artery fistulas. The occurrence o f transient wall motion abnormalities during high-dose dobutamine stress echocardiography in this patient showed that flow malperfusion may result in true ischemia during stress. Assessment o f myocardial ischemia with dobutamine stress echocardiography may be helpful in the evaluation o f anginal symptoms in patients with coronary artery fistulas. We are grateful to Jan Tuin and Ren~ Frowyn for the preparation of the figures.
REFERENCES
1. Libcrthson RR, SagarK, Berkoben JP, Weintraub ItaM,Levine FH. Congenital coronary arteriovenous fist-ulac. Circulation 1979;59:849-54. 2. Yamanaka O, Hobbs RE. Coronary artery abnormalities in 126,595 patients undergoing coronary angiography. Cathet Cardiovasc Diagn 1990;21:28-40. 3. AshrafSS,Shaukat N, Fisher M, ClarkeB, KeenanDJ. Bicoronary-pulmonaryfistulae with coexistent mitral valveprolapse: a case report and literature review of coronary-pulmonary fistula. Eur Heart I 1994;15:571-4. 4. Urmita-S CO, Fallaschi G, Ott DA, Cooley DE. Surgical management of 56 patients with congenital coronary artery fistulas. Ann Thorac Surg t983;35:300-7. 5. Said SA, Landman GH. Coronary-pulmonary fistula: Long term follow-up in operated and in non-operated patients. Int J Cardiol 1990;27:203-i0. 6. Brooks CH, Bates PD. Coronary artery-leftventricular fistula with angina pectoris. Am Heart J 1983;I06:404-6. 7. Glynn TP, Fleming RG, Haist ~L, Hunteman RK. Coronary arteriovenous fistula as a cause for reversible thallium-201 perfusion defect. J Nucl Med 1994;35:i808-10. 8. Said SA, Bucx JJ, van de Well FA. Stress MIBI scintigraphyin multiple coronary-pulmonaryfistula: failure to demonstrate 'steal' phenomenon. Int J Cardiol 1992;35:270-2. 9. SalustriA, Fioretti PM, Pozzoli MMA, McNeill AJ, Roelandt JRTC. Dobutamine stress echocardiography: its role in the diagnosis of coronary artery disease. Eur Heart I 1992;13: 70-7. 10. RuffoloRR. The pharmacologyofdobutamine. Am 5r Med Sci 1987;294:244-8.