Eur J Cardiothorac Surg 2008;33:939-941. doi:10.1016/j.ejcts.2008.01.048
Copyright © 2008, European Association for Cardio-thoracic Surgery. Published by Elsevier. All rights reserved.
Light-guided surgery to repair coronary sinus orifice atresia with left superior vena cava
Yukihiro Kaneko*,
Jotaro Kobayashi,
Yusuke Yamamoto,
Keiji Tsuchiya
Departments of Cardiovascular Surgery and Pediatrics, Japanese Red Cross Medical Center, 4-1-22 Hiroo, Shibuya-ku, Tokyo 150-8935, Japan
Received 5 November 2007;
received in revised form 15 January 2008;
accepted 30 January 2008.
* Corresponding author. Address: Department of Cardiovascular Surgery, Japanese Red Cross Medical Center, 4-1-22 Hiroo, Shibuya-ku, Tokyo 150-8935, Japan. Tel.: +81 3 3400 1311; fax: +81 3 3409 1604. (Email: yukihirokaneko{at}hotmail.com).
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Abstract
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An 8-month-old male with coronary sinus orifice atresia, left superior vena cava, and single ventricle underwent light-guided coronary sinus unroofing concomitant with bidirectional cavopulmonary anastomosis to circumvent coronary sinus hypertension. During surgery, a 2.25 Fr angioscopic catheter was inserted into the coronary sinus via the left superior vena cava. The coronary sinus, lit by the illumination obtained from the catheter, was readily located from the left atrial interior, and unroofed. Light-guided coronary sinus unroofing is an easy, safe, and quick technique for the creation of unobstructed coronary sinus drainage in patients with coronary sinus orifice atresia and left superior vena cava.
Key Words: Coronary sinus • Congenital heart defects • Cardiac surgery • Light-guided surgery
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1. Introduction
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Occlusion of the coronary sinus orifice associated with coronary venous drainage through the left superior vena cava (LSVC), namely, coronary sinus orifice atresia (CSOA) with LSVC, is an intrinsically benign but potentially hazardous cardiac anomaly. Coronary sinus hypertension caused by LSVC ligation or cavopulmonary connection may induce serious consequences [1–5]. The surgical creation of unobstructed coronary sinus drainage, that is to say, coronary sinus redirection, is advised in patients undergoing LSVC ligation or cavopulmonary connection [5–8].
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2. Case report
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A 0-day-old male was diagnosed on echocardiography as having double outlet right ventricle, ventricular septal defect, d-malposed great arteries, mitral atresia, hypoplastic left ventricle, hypoplastic aortic arch, and aortic coarctation. At age 9 days, an extended end-to-end aortic anastomosis and pulmonary artery banding were performed via a left thoracotomy. At age 5 months, CSOA with small LSVC was noted on angiography (Fig. 1
).
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Fig. 1. Anteroposterior view of an angiogram showing CSOA and LSVC. A white arrow indicates the atretic coronary sinus orifice. A bracket indicates the LSVC. Coronary venous drainage through the LSVC is seen.
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At age 8 months, surgery consisting of bidirectional cavopulmonary anastomosis (BCPA), atrial septal defect enlargement, and coronary sinus unroofing was done. Through a median sternotomy, cardiopulmonary bypass was established with aortic cannula, direct superior vena caval cannula, and inferior vena caval cannula advanced through the right atrium. An angioscopic catheter measuring 0.75 mm in diameter (Fiber catheter AS-003, FiberTech Inc., Tokyo, Japan) was inserted into the LSVC and advanced into the coronary sinus. With cardioplegic cardiac arrest, an oblique right atriotomy was made. After the atrial septal defect was enlarged, a dimple indicating an atretic mitral valve was exposed. The illumination from the tip of the angioscopic catheter was turned on, and the surgical light was dimmed. The illumination was clearly visible from the left atrial interior, and the location of the coronary sinus could be identified (Video 1). By moving the catheter back and forth the surgeon could clearly see the course of the coronary sinus. Under illumination from the catheter, the coronary sinus was incised from the left atrial interior using a stroker scalpel, and was then unroofed by extending the incision with scissors. The unroofing maneuver was completed in 3 min. The pulmonary trunk was divided, the LSVC was ligated, and the BCPA was made in the usual manner. Cardiopulmonary bypass time and aortic cross-clamp time were 100 min and 37 min, respectively. No arrhythmia or deterioration of ventricular function was noted postoperatively. The systemic ventricular diastolic diameter/systolic diameter on ultrasound study were 36.0 mm/29.3 mm before BCPA, and 34.0 mm/26.5 mm at 4 months after BCPA. The fractional shortening was 19.6% before BCPA, and 22.1% at 4 months after BCPA. The patient recovered uneventfully, and is awaiting a total cavopulmonary connection (TCPC).
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3. Comment
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In the published literature, 19 patients with CSOA and LSVC who had undergone cardiac surgery have been reported (Table 1
). Nine patients underwent LSVC ligation or right heart bypass without concomitant coronary sinus redirection that caused coronary sinus hypertension. Six of them experienced cardiac dysfunction. Coronary sinus hypertension was corrected in four of them, resulting in ameliorated cardiac function. Five patients underwent LSVC ligation or right heart bypass with concomitant coronary sinus redirection. Cardiac dysfunction was not reported in these patients.
The surgical techniques used for coronary sinus redirection in patients with CSOA and LSVC are shown in Table 1. The unroofing procedure, which was used in the majority of the reported cases, appears technically simple and quick. Excision of a coronary sinus orifice membrane is not applicable in patients with a long gap between the blind end of the coronary sinus and the right atrium [4]. The other two techniques are time consuming, and entail risks of suture constriction and suture line bleeding [1,5].
The left atrial interior is devoid of an anatomic landmark for the coronary sinus. Therefore, it is difficult to identify the coronary sinus when viewed from the left atrial interior. In all of the reported coronary sinus unroofing procedure for CSOA and LSVC, a probe was inserted into the LSVC, advanced into the coronary sinus, and then palpated to locate the coronary sinus. The probe must be large and stiff enough to be palpated in the presence of a surgical glove and the intervening left atrial musculature. Takabayashi and associates introduced a bougie as large as 2.5 mm in diameter into the coronary sinus. Nevertheless, they found it difficult to determine the location of the first left atrial incision [8]. The use of an oversized probe or overly forceful palpation may injure the coronary sinus. To overcome this shortcoming, instead of using a stiff probe and palpation, we used a flexible illuminating catheter and visual inspection to locate the coronary sinus. By visually locating the coronary sinus, the incision could be made accurately and quickly without unnecessary dissection. Therefore, injury to the circumflex artery or a transmural incision, which leads to bleeding, could be avoided.
We learned from the present case a small lesson concerning light-guided surgery. During the unroofing maneuver with the surgical light lit fully, the surgical field was too bright to identify the illumination from the angioscopic catheter. When the surgical light was totally turned off, the surgical field was too dark for optimal surgical maneuver. Our lesson is that the appropriate degree of surgical light attenuation should be tested and adjusted before surgery.
Fluorescence-guidance has recently been used to identify tumor tissue during surgical resection of malignant glioma. It has been shown that fluorescence-guidance enables a more complete resection and improves progression-free survival [9]. Light-guided intubation using a lighted stylet is used to manage intubation difficulties [10]. Thus, difficulty in identifying the target tissue during an intervention can sometimes be facilitated by visually controlled guidance. Given our experience, we advocate light-guided coronary sinus unroofing as an easy, safe, and quick coronary sinus redirection technique in patients with CSOA and LSVC.
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Appendix A
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Supplementary data
Supplementary data associated with this article can be found, in the online version, at doi:10.1016/j.ejcts.2008.01.048.
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Abstract
1. Introduction
