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Myocardial Ischaemia in a Case of a Solitary Coronary Ostium in the Right Aortic Sinus with Retroaortic Course of the Left Coron

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Myocardial Ischaemia in a Case of a Solitary Coronary Ostium in the Right Aortic Sinus with Retroaortic Course of the Left Coron Heart 1998;80:307–312 307 CASE REPORT Heart: first published as 10.1136/hrt.80.3.307 on 1 September 1998. Downloaded from Myocardial ischaemia in a case of a solitary coronary ostium in the right aortic sinus with retroaortic course of the left coronary artery: documentation of the underlying pathophysiological mechanisms of ischaemia by intracoronary Doppler and pressure measurements E R Schwarz, P K Hager, R Uebis, P Hanrath, H G Klues Abstract tional coronary obstruction and ischae- Only a few cases of a single coronary mia in this malformation. Bypass surgery ostium and retroaortic course of the was successfully performed with sympto- coronary artery have been described. matic improvement. Almost all cases reported so far had addi- (Heart 1998;80:307–312) tional coronary artery or valvar disease. However, myocardial ischaemia may be Keywords: coronary anomaly; Doppler; intravascular caused by the coronary malformation ultrasound; single coronary ostium; congenital disorders alone. A 40 year old woman with severe myocardial ischaemia in the absence of Med Clinic I, RWTH clinically relevant coronary atherosclero- University Hospital A solitary coronary ostium in or immediately sis is described. To clarify the origin and cephalad to the right aortic sinus and the Aachen, Germany mechanisms of ischaemia, intracoronary E R Schwarz course of the left coronary artery dorsal to the P K Hager Doppler, pressure and ultrasound studies ascending aorta is a very rare congenital http://heart.bmj.com/ P Hanrath were performed using microtransducers. anomaly. This coronary malformation has been H G Klues In its outer portion along the course classified as the type IV.A.2a according to the behind the ascending aorta, coronary 1 Medizinische Klinik, classification of Roberts. At present, only a few blood flow velocities were increased, there cases with this abnormality have been de- AschaVenburg, was an external elliptical compression, Germany scribed in the literature. In most of these and distal coronary flow reserve was R Uebis, patients, the diagnosis was incidental either reduced. Furthermore, an overshoot in during angiography or necropsy. Most patients Correspondence to: diastolic pressure above aortic pressure on September 29, 2021 by guest. Protected copyright. had significant concomitant coronary athero- Dr E R Schwarz, Heart was detectable within this portion. Dob- Institute Research, Good sclerosis causing symptoms of angina, or myo- utamine stimulation exaggerated the ob- Samaritan Hospital, 1225 cardial ischaemia and infarction.2–4 Even in the Wilshire Boulevard, Los served intracoronary haemodynamics absence of severe coronary atherosclerosis, Angeles, CA 90017–2395, and induced myocardial ischaemia. The USA. myocardial ischaemia can occur in cases of intracoronary diagnostic procedures per- anomalous origin of the left coronary artery formed were helpful in clarifying the Accepted for publication from the right sinus of Valsalva, if the left cor- 20 January 1998 pathophysiological mechanisms of func- onary artery passes posterior to the aorta.5 The pathophysiological mechanisms leading to myocardial ischaemia in the absence of coron- ary artery disease are incompletely understood. This is in contrast to other coronary anomalies where the left coronary artery arises from the right coronary artery and courses between the aorta and pulmonary artery (interarterial or intertruncal course). This has been associated with myocardial ischaemia and sudden death.6–10 The mechanism in this scenario is most likely a transient occlusion of the left cor- onary artery caused by an increase in blood flow through the aorta and the pulmonary artery, resulting in kinking or pinching of the Figure 1 (A) 12 lead ECG showing ST segment elevation in leads I, aVL, V2–V5 with artery.56 In contrast, in the abnormality ST segment depression in II and aVF directly after exercise testing, associated with severe symptoms of angina. (B) At rest, ST segment changes diminished and symptoms were presented here, coronary obstruction is usually alleviated. absent on angiography.11 308 Schwarz, Hager, Uebis Heart: first published as 10.1136/hrt.80.3.307 on 1 September 1998. Downloaded from Figure 2 Coronary angiogram ((A) left anterior oblique projection; (B) right anterior oblique projection) showing a solitary coronary ostium in the right aortic sinus and retroaortic course of the left coronary artery. Note the aorta (Ao) and the catheter in the pulmonary artery (PA). The arrow indicates the location of altered intracoronary haemodynamics compared with the proximal and distal segments, as confirmed by the position of the intravascular transducers angiographically. RCA, right coronary artery; LAD, left anterior descending coronary artery; CX, left circumflex coronary artery. We present the first report describing ured as the ratio of maximal flow velocity after myocardial ischaemia, and its potential patho- stimulation with 12 mg of intracoronary pa- physiological mechanisms, in a patient with a paverin to baseline flow velocity, was signifi- left coronary artery originating from the right cantly reduced at 1.7.17 sinus of Valsalva coursing posterior to the aorta Intracoronary pressure measurements dem- in the absence of clinically relevant coronary onstrated a local overshoot in diastolic pressure atherosclerosis. (fig 5) at the site of accelerated flow velocity within the course of the left descending coron- Case report ary artery. In vitro validation of pressure meas- A 40 year old woman reported a four week urements within squeezed coronary segments using the 0.014 inch pressure microtransducer history of exercise induced angina pectoris; ST 18 http://heart.bmj.com/ segment elevation was documented during has recently been demonstrated. Intravascu- bicycle ergometry in the anterior electrocar- lar ultrasound demonstrated only a slight diographic leads (V2–V5) (fig 1). Immediately systolic and diastolic reduction of cross sec- after exercise testing, torsade de pointes tional area at this portion (fig 4A) compared occurred. There was no history of coronary with a normal distal portion (fig 4B), without artery disease or hypertension. The patient signs of atherosclerosis. Measurements during had a history of cigarette smoking for a few intravenous infusion of dobutamine demon- years before admission. Physical examination strated augmentation of blood flow velocities revealed an otherwise healthy woman. Coron- and of intravascular pressure (fig 5), and a on September 29, 2021 by guest. Protected copyright. ary angiography revealed a solitary ostium in further reduction in cross sectional area. the right aortic sinus associated with an aber- Moreover, angina symptoms with ST segment rant retroaortic course of the left coronary elevation in the anterior leads occurred similar artery (figs 2 and 3). There was a 30% athero- to those during exercise testing. In view of the sclerotic stenosis in the mid-portion of the severity of symptoms and objective evidence of right coronary artery. Despite several addi- tional angiographic projections, it was impos- sible to demonstrate a significant phasic lumi- LCA nal narrowing of the left main within its AO retroaortic course. Intracoronary Doppler flow velocity and pressure measurements using miniaturised 0.014 inch guidewire transducers and intravas- CX cular ultrasound were performed at rest and during dobutamine stimulation.12–16 Intravas- RCA cular Doppler showed only slightly increased blood flow velocities along the curved retroaor- PA tic segment of the left main stem (arrow, fig 3), LAD which were enhanced during intravenous infu- sion of dobutamine (5–20 µg/kg) (fig 4A), Figure 3 Schematic drawing of the coronary anomaly compared with measurements at rest (fig 4B) with the course of the left coronary artery posterior to the and with measurements at the proximal or dis- ascending aorta. AO, aorta; PA, pulmonary artery; LCA, left coronary artery; RCA, right coronary artery; CX, left tal portions of the left coronary artery (not circumflex coronary artery; LAD, left anterior descending shown). Distal coronary flow reserve, meas- coronary artery. Microtransducer diagnostics in coronary malformation 309 Heart: first published as 10.1136/hrt.80.3.307 on 1 September 1998. Downloaded from Figure 4 Intracoronary blood flow velocities (left) and intravascular ultrasound (right) within the left coronary artery at its sharp course dorsal the ascending aorta during dobutamine stimulation (A) and at rest (B). ADPV,average diastolic flow velocity; ASPV,average systolic flow velocity; APV,average peak flow velocity; DSVR, diastolic/systolic velocity ratio; BAPV,basic average peak flow velocity; PAPV,peak average peak flow velocity. RATIO, coronary flow reserve. http://heart.bmj.com/ on September 29, 2021 by guest. Protected copyright. Figure 5 Intracoronary pressure measurements, obtained using a fibreoptic microtransducer incorporated in the distal segment of a 0.014 inch guidewire, show the overshoot in pressure within the retroaortic course of the left coronary artery compared with aortic pressure. To avoid artificial measurements, the guidewire was repeatedly rotated and its position angiographically confirmed. There was no visible external compression causing catheter entrapment. LCA, left coronary artery. ischaemia and ischaemia induced malignant Discussion arrhythmias, the patient had aortocoronary Congenital coronary malformations are rare bypass surgery with the left internal mammary causes of angina, myocardial infarction or artery to the left anterior
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