International Journal of Cardiology 184 (2015) 296–298

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International Journal of Cardiology journal homepage: www.elsevier.com/locate/ijcard

Letter to the Editor

A case of cardio-pulmonary arrest caused by anomalous origin of left main coronary artery from right sinus of valsalva Masayoshi Kiyokuni a,⁎, Motohiko Goda c, Makoto Okiyama d, Chika Kawashima b, Hiroshi Doi b, Akio Hisa b, Sakie Tomari b, Takayuki Mitsuhashi b, Hideyuki Iwaki d, Tsutomu Endo b, Satoshi Umemura a, Munetaka Masuda c a

Department of Medical Science and Cardiorenal Medicine, Yokohama City University School of Medicine, Yokohama, Japan Division of Cardiology, Saiseikai Yokohama City Southern Hospital, 3-2-10 Konandai, Konan Ward, Yokohama, Japan c Department of Surgery, Yokohama City University School of Medicine, Yokohama, Japan d Division of Surgery, Saiseikai Yokohama City Southern Hospital, 3-2-10 Konandai, Konan Ward, Yokohama, Japan b

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Article history: Received 10 January 2015 Accepted 21 February 2015 Available online 24 February 2015 Keywords: Coronary vessels Anomalous origin Myocardial ischemia Chest pain Cardio-pulmonary arrest

A 15-year-old boy experienced chest discomfort after playing football. Emergency medical service was activated and he was transported to our hospital. At the time of admission, he went into cardiopulmonary arrest. Because he had experienced several episodes of chest discomfort during effort beginning one year prior to this episode, he had undergone echocardiography and a treadmill exercise test at another hospital one week prior to cardio-pulmonary arrest. At that time, his cardiac function had been good and he had shown no signs of hypertrophy or abnormal shunting. The treadmill exercise test had revealed significant STsegment depression in leads aVF, V5 and V6 when the patient's heart rate was greater than 180 beats/min, although no symptoms were observed. No further examination or medical therapy had been performed. He had no relevant medical history or family history. He had not been ill prior to his episode of cardio-pulmonary arrest and had no allergies and no history of smoking or drug use. Immediately after admission, advanced cardiac life support was performed. An electrocardiogram showed ventricular fibrillation. After a single DC shock administered via a biphasic defibrillator, his heart ⁎ Corresponding author at: Department of Medical Science and Cardiorenal Medicine, Yokohama City University School of Medicine, Yokohama 236-0004, Japan. E-mail address: [email protected] (M. Kiyokuni).

http://dx.doi.org/10.1016/j.ijcard.2015.02.069 0167-5273/© 2015 Elsevier Ireland Ltd. All rights reserved.

returned to sinus rhythm. His Glasgow coma scale score after the shock was 12 (E3V4M5) and he was intubated with appropriate anesthesia. The other vital signs were as follows: blood pressure: 84/ 40 mm Hg; heart rate: 130 beats/min; and body temperature: 36 °C (96.8 °F). A chest exam revealed a galloping rhythm of the heart sounds with no murmur and decreased breath sounds with bilateral coarse crackling, indicating acute pulmonary edema. The other physical examinations revealed no abnormalities. Electrocardiogram after the cardioversion showed global ischemia with QRS duration N120 ms, STsegment elevation N 1 mm in lead aVR and ST-segment depression N2 mm in leads aVF, V5 and V6. Echocardiography showed akinesis of the anterolateral wall with 20% ejection fraction and no valvular disease or pericardial effusion. Chest radiography also showed pulmonary edema with a cardio-thoracic ratio of 47%. We diagnosed the patient with acute myocardial infarction. We immediately performed coronary angiography, which was started within 30 min after admission. Although we tried several standard techniques, we could not engage the left main coronary artery (LMCA). The right coronary artery was normal. Finally, aortography revealed that the ostium of the LMCA had 90% stenosis, and the Thrombolysis In Myocardial Ischemia (TIMI) flow grade was 3. As the patient's vital signs were stable, we decided not to treat his stenosis by stent implantation during this acute phase; instead, he was transferred to the cardiac care unit. Peak creatinine kinase was 7294 IU/L. With the exception of the cardiac enzymes, the other laboratory tests were within normal limits. On the third day after admission, a contrast-enhanced coronary computed tomography (CT) angiography showed that his LMCA arose from the right sinus of valsalva (RSOV) and coursed between the aorta and the pulmonary artery. Two severe stenoses existed in the anomalous LCMA. One was a slit-like stenosis in the LMCA ostium; the other was a severely stenotic region with acute angulation in the distal portion of the LMCA near the bifurcation of the left descending artery (LAD) and the left circumflex artery (LCx) (Fig. 1). After the CT angiography, the patient went into cardio-pulmonary arrest again. He received percutaneous circulatory support (PCPS) associated with intra-aortic balloon pumping (IABP). As on admission day, aortography at this time revealed that the ostium of the LMCA had 90% stenosis and that the TIMI flow grade was 3. On the fourth day

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(a) 1)

2) PA

PA Ao

R

R

LA

R

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4)

RCA

LMCA

PA

PA

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Ao LA

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Fig. 1. Coronary CT angiography image during the acute phase demonstrated a slit-like stenosis in the LMCA ostium; there was also a severely stenotic region with acute angulation in the distal portion of the LMCA near the bifurcation of the LAD and the LCx. Transverse CTA image (a), and curved planar reconstruction coronary CTA image (b). LMCA, left main coronary artery; RCA, right coronary artery; LAD, left anterior descending coronary artery; LCx, left circumflex artery; HL, high lateral; PA, pulmonary artery; Ao, aorta; LA, left atrium.

after admission, as the patient's cardiac function remained severely depressed in spite of circulatory support, emergent coronary artery bypass graft (CABG) was performed, grafting the left internal thoracic artery to the LCx and the right internal thoracic artery to the LAD. During this

operation, the slit-like ostial stenosis and the severely stenotic region in the distal portion of the LMCA were confirmed by transesophagus echocardiography (Fig. 2). Cardioplegic cardiac arrest to confirm both intra-mural running and ostial stenosis of LMCA was avoided because

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a)

b) Commissure

Ao Ao

LCx

HL LMCA ostium PA

PA LAD

Fig. 2. Transesophagus echocardiography revealed two stenotic regions in the LMCA: a slit-like ostial stenosis (yellow arrow) and an acute angulation with stenosis after the split from the aorta (red arrow).

additional ischemia might have resulted in irreversible cardiac damage. Three days after CABG, the patient was successfully weaned from PCPS; he remained on IABP for an additional three days. On the 35th day after admission, coronary angiography showed that both bypass grafts were patent, yet flow competition against native coronary flow was also observed. After confirmation that no significant ischemic ST-changes or symptoms occurred during around 7 METs of exercise, the patient was discharged on the 38th day after admission and is still doing well eighteen months after the surgery without additional intervention. Discussion Coronary artery anomalies are very rare, occurring in less than 1% of the general population, and are not well understood. Angelini et al. have reported the incidence, pathophysiology, and clinical relevance of coronary artery anomalies [1]. In the present case, the LMCA arose from the RSOV, then took an anomalous course between the aorta and the pulmonary artery; this type of anomaly is called interarterial direction [2]. To our knowledge, this is the first report of a coronary artery anomaly patient with the LMCA arising from the RSOV who presented with cardio-pulmonary arrest, was treated by CABG, and successfully recovered. Very few cases of LMCA stenting for this type of anomalous coronary artery have been reported previously, and its long-term prognosis remains unknown [3]. Surgery is likely to be indicated in symptomatic patients with this anomaly. Surgical options are as follows: (a) unroofing the LMCA ostium, (b) trans-locating the LMCA from the right sinus to the left sinus, (c) coronary artery bypass grafting and (d) mobilization of the pulmonic root [4]. In this case, neither unroofing nor coronary artery translocation nor mobilization of the pulmonary artery could be selected because two organic stenoses existed. The LMCA ostium had a slit-like stenosis; there was also a severely stenotic region with acute angulation of the distal portion of the LMCA near the bifurcation of the

LAD and the LCx. These stenoses were observed by both coronary CT angiography and transesophagus echocardiography. Accordingly, we selected CABG as the treatment method for this patient. It is important to consider that the long-term prognosis of patients treated by CABG is unclear. Because the patient's native coronary blood flow during rest was scored at a TIMI flow grade of 3, as observed in both the emergency coronary angiography and a second coronary angiography after CABG, blood flow competition may occur between the native flow and the bypass graft. The long-term patency of bypass grafts is doubtful, and intensive clinical follow-up will be necessary for this patient. Re-operation for an eradicative treatment may be needed in the future. Conflict of interest No conflict of interest exists. Informed consent The patient and his parents have given their informed consent for this case report to be published. References [1] P. Angelini, J.A. Velasco, S. Flamm, Coronary anomalies: incidence, pathophysiology, and clinical relevance, Circulation 105 (20) (2002) 2449–2454. [2] J. Ni, C. Ge, P. Wang, et al., Undescribed origin of the left main coronary artery from the right sinus of valsalva: an explanation of nonexistent myocardial ischemia? Int. J. Cardiol. 174 (2) (2014) e32–e33. [3] R. Jaffe, A. Shiran, T. Gaspar, et al., Primary stenting of an anomalous left main coronary artery with an interarterial course during cardiac arrest: imaging with CT angiography, Circ. Cardiovasc. Imaging 2 (4) (2009) 351–352. [4] K. Nimdet, J. Soongswang, S. Sriyodchati, An interarterial course of anomalous coronary artery and an alternative surgical option, Cardiol. Young 22 (5) (2012) 536–538.

A case of cardio-pulmonary arrest caused by anomalous origin of left main coronary artery from right sinus of valsalva.

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