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Ann Thorac Surg 1995;60:84-89
© 1995 The Society of Thoracic Surgeons

Aortic Implantation Is Possible in All Cases of Anomalous Origin of the Left Coronary Artery From the Pulmonary Artery

California Pacific Medical Center, Kaiser Permanente Medical Center, and University of California, San Francisco, California

	Abstract

Top Footnotes Abstract Introduction Material and Methods Results Comment Acknowledgments References

 
Background. Anomalous origin of the left coronary artery from the pulmonary artery (PA) optimally is treated by creation of a multiple coronary system. This study explores the use of aortic implantation employing alternative methods to achieve coronary transfer in all patients, regardless of the site of origin of the anomalous coronary artery, avoiding the problems of bypass grafts and tunnel procedures.

Methods. During the period 1986 to 1994, 11 patients aged 6 months to 8 years (mean age, 2.6 years) underwent repair. Coronary artery origin from the PA included left sinus in 3, posterior in 2, right sinus in 2, intramural aorta with its orifice at the bifurcation of the main and right PA in 1, high left main PA in 1, high at the bifurcation of main and right PA in 1, and anterior in 1. Findings included angina in 4, prior infarctions in 3, ischemia in 7, left ventricular dysfunction in 6, mitral regurgitation in 5, atrial septal defect in 2, and echocardiograms suggestive of endocardial fibrosis in 4. One patient had prior ligation with ventricular dysfunction and collateralization and recanalization. A single patient was asymptomatic. Repair was accomplished by direct transfer using the PA sinus of Valsalva as a button in only 6; tubular reconstruction was used in 4 when the distance was too great to avoid tension; 2 short tubes were constructed with PA wall in 2 of the 3 left sinus origins, whereas 2 long tubes of PA wall were used (1 high on the left side of the main PA and 1 with left anterior descending origin from the anterior sinus of Valsalva in a patient with malrotation [end neo-artery to side aortic reconstruction]); finally, in situ transfer and intraaortic reconstruction (unroofing and anastomosis) was performed in 1 intramural coronary artery. Division of the PA, mobilization of the distal PA, division of the ductus, and direct reanastomosis of the PA was performed in 3 tubular reconstructions, as well as all 6 direct coronary transfers.

Results. There were no operative or late deaths. Follow-up of 2 to 100 months (mean, 46 months) revealed no new angina or infarctions, improved function and decreased mitral regurgitation. Echocardiographic and angiographic studies demonstrated patency and prograde flow in the new coronary systems.

Conclusions. Aortic implantation is the treatment of choice for anomalous origin of the left coronary artery. Methods such as direct transfer, tubular reconstruction, and in situ transfer make such implantation possible in all patients regardless of the site of coronary origin, distance from the aorta, or coronary artery configuration.

	Introduction

Top Footnotes Abstract Introduction Material and Methods Results Comment Acknowledgments References

 
See also page 89.

Anomalous origin of the left coronary artery from the pulmonary artery is a rare congenital anomaly that often presents insidiously in early infancy with infarction. First described in 1885, the clinical syndrome was explored by Bland, White, and Garland in 1933 [1]. They demonstrated the combined effects of absence of a normal coronary flow with a coronary steal and the profound ischemia that can result. Early attempts to treat this problem with a simple ligation resulted in a high mortality, and construction of a two coronary artery system has been recognized as the treatment of choice [2, 3]. The optimal method of such reconstruction remains controversial. Methods used in the past to achieve this goal include bypass graft procedures with the potential for distal anastomotic stenosis and tunnel procedures, in which tunnel obstruction and pulmonary arterial obstruction have been reported [2–8]. Coronary transfer, popularized by experience with neonatal arterial switch procedures, currently represents the most attractive method of creating a multiple coronary system and has been employed successfully in many such patients [3, 9–12]. However, anatomic variations of the anomalous coronary origins often preclude direct transfer [3, 12]. The current study explores methods of aortic implantation including both standard transfer techniques and alternate methods of arterial transfer regardless of the site of origin of anomalous coronary to provide arterial reconstruction of a multiple coronary artery system in all patients.

	Material and Methods

Top Footnotes Abstract Introduction Material and Methods Results Comment Acknowledgments References

 
Patients
During the period 1986 to 1994, 11 patients, aged 6 months to 8 years (mean age, 2.6 years) underwent aortic implantation. The site of coronary artery origin from the pulmonary artery included the left sinus of Valsalva in 3 patients, posterior in 2 patients, right sinus of Valsalva in 2 patients, intramural aorta with orifice at the bifurcation of the main and right pulmonary artery in 1 patient, high on the left main pulmonary artery in 1 patient, high at the bifurcation of the main pulmonary artery and right pulmonary artery in 1 patient, and anterior or facing sinus in 1 patient (the left anterior descending coronary in a patient with malrotation) (Fig 1). The pulmonary sinuses were designated as right, left, and facing according to the view from the posterior commissure looking anteriorly.

View larger version (21K): [in this window] [in a new window]   Fig 1. . Site of coronary origin from the pulmonary artery (PA) in the current series: (a) right sinus of Valsalva in 2, (b) left sinus of Valsalva in 3, (c) anterior in 1 (malrotation of left anterior descending coronary artery), (d) posterior in 2, (e) left main pulmonary artery high in 1, and (f) right main pulmonary artery at bifurcation of the right pulmonary artery in 2. (Ao = aorta.)

Surgical Technique
Operation for all patients was performed via a median sternotomy and bicaval cannulation with cardiopulmonary bypass with moderate hypothermia. Both great arteries then were clamped in 7 patients with cold blood cardioplegic solution administered both to the aortic root and main pulmonary artery. In 4 patients aortic root administration alone occurred with digital occlusion of the anomalous coronary orifice. In the most recent cases, continuous retrograde cardioplegia has been added during the procedure. In addition venting of the left heart structures via the atrial septum, left atrial appendage, or right superior pulmonary vein was used to prevent distention in these compromised ventricles. The surgical technique involved division of the main pulmonary artery and mobilization of the distal pulmonary artery with division of the ductus in 10 of the 11 patients. In 6 of these patients the ostium of the left coronary artery was excised with a sizeable cuff of adjacent sinus of Valsalva and the left coronary artery was mobilized. A punch then was used to produce an opening in the left lateral side of the ascending aorta, care being taken to avoid injury to the aortic valve by a low incision. The anastomosis was performed using 7-0 absorbable continuous suture (polyglyconate) (Fig 2). In 4 patients, tubular reconstruction was used when the distance was too great to avoid tension. Two short tubes were constructed from the pulmonary artery wall in 2 of the 3 patients with a left sinus of Valsalva origins of the anomalous coronary to avoid such tension. Two short lateral flaps were dissected and lateral suture lines constructed, again using absorbable 7-0 suture material. The proximal anastomosis was performed as with the standard direct transfer (Fig 3). In 1 patient the origin was high on the left side of the main pulmonary artery. In this case two long flaps of main pulmonary artery tissue were used to construct a long tube. This tube was then anastomosed to the aorta, again with 7-0 absorbable suture (Fig 4). In 1 patient with malrotation a left anterior descending coronary artery arose from the anterior or facing sinus of Valsalva (Fig 5); a long flap of pulmonary artery wall based to the left was used to fold around the orifice of the left anterior descending coronary and a single suture line of 7-0 absorbable suture was constructed to produce a tube, which then was anastomosed to the anterior aspect of the ascending aorta. Finally, in 1 patient, the coronary artery passed behind the main pulmonary artery and entered the aortic wall, running intramurally, and then exited to find its origin at the bifurcation of the main and right pulmonary artery (Fig 6). In this case the pulmonary artery was not divided, but rather the aorta was incised. The intramural portion of the coronary was unroofed intraluminally, and its intima was attached to the aortic intima with interrupted 7-0 absorbable suture. The origin of the coronary artery at the main to right bifurcation then was ligated. In this case the aorta was closed directly and the cross-clamp removed. In the 10 patients in whom division of the pulmonary artery was performed, the cross-clamp was removed after completion of the coronary anastomosis to evaluate coronary perfusion. Main pulmonary artery continuity then was reestablished by direct anastomosis in 9. This was possible due to the aggressive mobilization of the distal pulmonary artery and division of the ductus as performed in mobilization for the LeCompte maneuver. In the single patient with the left anterior descending coronary arising anteriorly, a pericardial patch was used to reconstruct the longitudinal flap defect produced by mobilization of tissue for the tube reconstruction (see Table 1).

View larger version (16K): [in this window] [in a new window]   Fig 2. . Coronary transfer. A cuff of pulmonary arterial wall is excised with the orifice of the aberrant coronary and the artery is mobilized distally. A punch is used to create an orifice in the aorta and anastomosis is performed. The main pulmonary artery, having been dissected to the hilum bilaterally and the ligamentum divided, then is anastomosed in an end-to-end fashion.

View larger version (16K): [in this window] [in a new window]   Fig 3. . Short tube reconstruction used in 2 patients.

View larger version (21K): [in this window] [in a new window]   Fig 4. . Long tube reconstruction with two lateral flaps used to construct the tube from high on the left side of the main pulmonary artery.

View larger version (18K): [in this window] [in a new window]   Fig 5. . Long tube reconstruction of the left anterior descending coronary artery arising anterior in a patient with malrotation. The orifice was in the sinus of Valsalva and eccentrically placed. Thus, the entire sinus was excised with a long flap of tissue superiorly. This was folded over with a single suture line laterally. The pulmonary artery was reconstructed using autologous pericardium.

View larger version (21K): [in this window] [in a new window]   Fig 6. . Unusual path of the intramural coronary artery with its origin at the bifurcation of the main and right pulmonary artery. Reconstruction was accomplished by unroofing the intramural portion of the coronary within the aorta, suturing the coronary intima to the aortic intima, and ligating the orifice of the aberrant coronary artery.

	Results

Top Footnotes Abstract Introduction Material and Methods Results Comment Acknowledgments References

 
There were no early or late deaths during the follow-up of 2 to 100 months (mean, 46 months). Echocardiographic evaluation was available in 11 and angiographic evaluation in 4. In each case, patency and prograde flow of the new coronary artery systems were demonstrated and no pulmonary artery obstructions were evident. No new angina has occurred and no new infarctions were demonstrated by electrocardiography. Improved left ventricular function was noted in 5 of 6 patients, although this became evident 3 to 6 months postoperatively in 2. Mitral regurgitation was improved in 4 of 5 patients with only mild insufficiency persistent in a patient with echocardiogram suggestive of endocardial fibroelastosis. Of the nine direct anastomoses, four demonstrated no valve insufficiencies and therefore right ventricular/pulmonary artery pressure could not be estimated. In 3 patients normal right ventricular/pulmonary artery pressures of 15, 12, and 14 mm Hg were observed. In the most recent patient a pulmonary artery gradient of 22 mm Hg was noted on initial postoperative examination, which has decreased to 16 mm Hg 6 months postoperatively. In 1 patient a 34 mm Hg estimated right ventricular pressure was recorded with a pulmonary artery gradient of 16 mm Hg. The right ventricular pressure decreased to 30 mm Hg 1 year later with gradient unchanged (mild stenosis). No residual shunts were noted in the patients with atrial septal defects, and reversal of the flattened septal motion in these patients was indicative of atrial septal closure rather than specific reversal of ischemia. Echocardiogram suggestive of endocardial fibroelastosis persisted in all 4 patients in whom it had been present preoperatively.

	Comment

Top Footnotes Abstract Introduction Material and Methods Results Comment Acknowledgments References

 
This study explores the methods available to create aortic implantation in all patients with anomalous left coronary artery systems from the pulmonary artery. Backer and associates [3], Alexi-Meskishvili and co-workers [12], and Vouhé and colleagues [10] clearly have demonstrated the efficacy of the creation of a multiple coronary system in such patients, and experience with arterial transfer in patients with transposition of the great arteries has made this modality standard for repair of the anomalous coronary artery. Other methods such as bypass grafting and tunnel procedures have been restricted to difficult sites of origin. Each of these methods is fraught with potential late complications, in the former distal anastomotic stenosis with obstruction and occlusion and in the latter, both obstruction of the tunnel itself and distal coronary occlusion, as well as pulmonary arterial obstruction [2, 7, 8]. Repair using the method described in our 5 patients in whom direct transfer was not possible affords alternatives to these methods avoiding their potential shortfalls. Armer and associates [13] first described the flap reconstruction approach in an autopsy reconstruction in 1965. We described our preliminary experience with this approach at the meeting of the European Chapter of the International Society for Cardiovascular Surgery in 1992. Subsequent to this report Tashiro and associates [14] described 2 adult patients in whom a tubular reconstruction was used with a side-to-side anastomosis posterior to the aorta. This technique, although providing an extravascular tunnel, has the potential for tunnel obstruction seen in the Takeuchi technique, because a side-to-side anastomosis with blind end and large saccular component results. The direct tubular approach used in the current study provides for linear flow from the aortic lumen to the distal coronary artery.

In the current study no patient required delayed sternal closure or ventricular assist. The absence of a patient with acute infarction in the study group accounts in part for this, but also close observation to the principles of myocardial protection via both antegrade and retrograde cardioplegic administration, attention to adequate perfusion by prevention of pulmonary arterial run-off, and protecting the left heart structures from acute distention [10, 12]. Improvement in ventricular function has been demonstrated after reestablishment of normal coronary perfusion [10, 12, 15]. This may be present initially or may be delayed for several months.

Although the Berlin group recommends a modified Kaye annuloplasty to improve mitral valve function, this was performed only in severe class 3 mitral incompetence in that series [12]. In the current series only mild to moderate regurgitation was noted and the response to myocardial revascularization, even in the face of endocardial fibroelastosis, produced excellent hemodynamics and avoided the need for mitral valve intervention.

Finally, echocardiogram suggestive of fibroelastosis persisted in 4 patients, consistent with prior experience, reinforcing an aggressive approach to the identification and treatment of this anomaly at the earliest possible time [16]. It was hoped with the advent of color-flow Doppler two-dimensional echocardiography that early recognition of this problem could be achieved before infarction. However, recent experience has tempered our enthusiasm and must heighten our vigilance in identifying this insidious problem so that repair can occur before devastating infarction.

The current approach represents such a method of repair. It can provide complete arterialization of a multiple coronary system, while avoiding the inherent problems associated with bypass and tunnel procedures. The method employs pulmonary arterial wall as in coronary transfers, where the cuff of the sinus of Valsalva is used to facilitate the transfer. No evidence of aneurysm formation has been encountered in using this technique in either the arterial switch or transfers in anomalous coronary arteries such as seen in this series. Thus, both short and long tubular reconstructions, which use the same material constructed into tubular extravascular structures providing linear flow from the aorta to the distal pulmonary artery bed, should provide a similar conduit. Care has been taken in the initial construction of these conduits to avoid any saccular configuration as in the Takeuchi or Tashiro procedure, and the approach itself avoids the potential for distal anastomotic obstruction inherent to bypass procedures.

Aortic implantation is the treatment of choice for anomalous origin of the coronaries. Methods such as direct transfer, tubular reconstruction, and in situ transfer make such implantations possible in all patients regardless of the site of coronary artery origin, distance from the aorta, or coronary artery configuration.

	Acknowledgments

Top Footnotes Abstract Introduction Material and Methods Results Comment Acknowledgments References

 
We thank Sophia E. Seto for research and preparation of the manuscript.

	Footnotes

Top Footnotes Abstract Introduction Material and Methods Results Comment Acknowledgments References

 
Presented at the Thirty-First Annual Meeting of The Society of Thoracic Surgeons, Palm Springs, CA, Jan 30–Feb 1, 1995.

Address reprint requests to Dr Turley, Pediatric Cardiovascular Surgery, California Pacific Medical Center, 2100 Webster St, #332, San Francisco, CA 94115.

	References

Top Footnotes Abstract Introduction Material and Methods Results Comment Acknowledgments References

 

1. Bland EF, White PD, Garland J. Congenital anomalies of the coronary arteries. Am Heart J 1933;8:787–801.
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3. Backer CL, Stout MJ, Zales VR, et al. Anomalous origin of the left coronary artery. A twenty-year review of surgical management. J Thorac Cardiovasc Surg 1992;103:1049–58.[Abstract]
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9. Neches WH, Mathews RA, Park SC, et al. Anomalous origin of the left coronary artery from the pulmonary artery: a new method of surgical repair. Circulation 1974;50:582–7.[Abstract/Free Full Text]
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13. Armer RM, Shumaker HB, Lurie PR, Fisch C. Origin of the left coronary artery from the pulmonary artery without collateral circulation: report of a case with suggested surgical correction. Pediatrics 1965;32:588–93.[Abstract/Free Full Text]
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This Article Abstract Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Kevin Turley Robert J. Szarnicki Keith D. Flachsbart Richard C. Richter Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Turley, K. Articles by Tarnoff, H. Search for Related Content PubMed PubMed Citation Articles by Turley, K. Articles by Tarnoff, H. Related Collections Related Article