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From the *Department of Anesthesiology, Osaka Medical College, Takatsuki, Japan, and Emergency and Critical Care Medicine, The University of Tokushima Graduate School, Tokushima, Japan.
Address correspondence and reprint requests to Toshiyuki Sawai, MD, PhD, Department of Anesthesiology, Osaka Medical College, 2–7 Daigaku-machi, Takatsuki 569-8686, Japan. Address e-mail to ane026{at}poh.osaka-med.ac.jp.
A 35-yr-old healthy woman was admitted for evaluation of an abnormal cardiac mass on two-dimensional transthoracic echocardiography (TTE). TTE demonstrated an abnormal giant mass dorsad to the right atrium and right ventricle. Although color Doppler TTE revealed abnormal blood flow within the left atrium (LA), the mass was not well visualized. Coronary angiography revealed a normal left coronary artery and an 80-mm internal diameter (ID) aneurysm at the proximal portion of right coronary artery (RCA). The giant aneurysm did not allow the distal part of RCA to be visualized. Meanwhile, three-dimensional computed tomography (3-D CT) revealed three giant coronary artery aneurysms (CAAs) (Fig. 1). To avoid the risk of aneurysm rupture, surgery was planned.
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After induction of general anesthesia, a transesophageal multiplane probe (Philips Electronics, Eindhoven, Netherlands) was placed. A midesophageal four-chamber-view confirmed that a CAA compressed the right atrium and right ventricle, and color-Doppler imaging suggested abnormal blood flow draining into the LA (Fig. 2A) (Video Clip 1; please see video clip available at www.anesthesia-analgesia.org). Both ventricles had normal dimensions, good contractility, and no abnormalities in regional wall motion. Similar to the image plane on the 3-D CT, a transesophageal electrocardiography (TEE) view from midesophagus (37 degree multiplane view) revealed that the three CAAs lay in series, with an abnormal duct connecting the first (ID 80 mm) to the other 2 (ID 25 and 20 mm). In addition, abnormal turbulence was detected around the third CAA. By rotating the TEE probe left at approximately the same depth, we identified the coronary artery fistula draining into the LA (Fig. 2B) (Video Clip 2; please see video clip available at www.anesthesia-analgesia.org).
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After establishing cardiopulmonary bypass, the aorta was opened and a cardioplegia cannula in the coronary ostia used to obtain cardiac arrest. Figure 3 schematically outlines the CAAs and the coronary artery fistula. Figure 4 shows a photograph taken during the intraoperative period. We closed the coronary artery fistula, bypassed the distal RCA to the aorta with a saphenous vein graft, and removed all CAAs. There was no thrombus in any of the aneurysms. Subsequent TEE confirmed that the flow through the coronary artery fistula was no longer present and that ventricular contractility was normal. Postoperative recovery was uneventful, and she was discharged from the hospital on chronic anticoagulation therapy.
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The incidence of CAA with fistulous formation is 0.2%.1 They most frequently occur around the atria. A diameter larger than 50 mm in adults is considered as "giant," and is usually due to Kawasaki disease. Most of the previously reported cases with fistula are congenital.1 When present, the fistula usually drains into the right atrium or right ventricle, and only rarely into the LA.1 Multiple giant CAAs coexistent with fistulous drainage, as in this patient, are very rare. In cases where CAA and fistula coexist, the risk of aneurysmal rupture increases with older age (especially older than 40 yr), angina (older than 50 yr), congestive heart failure, infectious endocarditis, distal embolization, and other complications.2 In our patient, complications were not observed, probably because she was young, the RCA distal to the CAA was patent, and the effect of left-to-left shunt due to the fistula was negligible.
To minimize surgical risk, precise information about fistulous drainage is usually obtained by coronary catheterization.3 However, intraoperative TEE adds more information than coronary catheterization, which could not show the drainage site of the fistula.3 In this case, dilution of the contrast medium and overlapping of adjacent structures made detailed characterization difficult, whereas the combined use of intraoperative TEE, preoperative TTE, and 3-D CT facilitated detection of the fistulous drainage. Because the LA is acoustic window for TEE, we could identify coronary fistulous flow as an abnormal flow, and characterize the origin, course, and drainage site. This information helped the surgeon to identify the drainage site of the fistula. Surgical repair of fistula does not always require cardiopulmonary bypass4 and endovascular therapy using transcatheter coil has been successfully used.5 We decided, however, to use complete cardiopulmonary bypass because the fistulous tract was not visible from the surface of the beating heart and removal of all the CAAs was deemed necessary. Because TEE indicated that the fistula was present between the LA and the RCA, we chose individual perfusion of cardioplegia to obtain cardiac arrest. If there had been no information about the site of the drainage of the coronary artery fistula, retrograde cardioplegia perfusion might have been required to obtain cardiac arrest. Postrepair TEE was also useful in confirming the disappearance of the fistulous flow.
In conclusion, intraoperative TEE was useful in identifying the precise location of the CAAs and the CAF. Postoperative TEE was also useful in confirming the absence of the CAF and normal ventricular wall motion.
Footnotes
This article has supplementary material on the Web site:www.anesthesia-analgesia.org.
Accepted for publication November 30, 2007.
REFERENCES
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