Coronary Arterial Anatomy in Double-Outlet Right Ventricle With Subpulmonary VSD

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1 Coronary Arterial Anatomy in Double-Outlet Right Ventricle With Subpulmonary VSD Hideki Uemura, MD, Toshikatsu Yagihara, MD, Yasunaru Kawashima, MD, Kyoichi Nishigaki, MD, Tetsuro Kamiya, MD, Siew Yen Ho, PhD, and Robert H. Anderson, MD National Heart and Lung Institute, London, United Kingdom, and National Cardiovascular Center, Osaka, Japan We have examined 38 hearts with a double-outlet right ventricle with a subpulmonary ventricular septal defect. We divided the hearts into three groups according to the angle between the planes formed between the outlet septum and the remainder of the muscular ventricular septum; namely, at approximately right angles (15 hearts), parallel (11 hearts), and at an acute angle (12 hearts). The coronary arterial pattern corresponding to that seen in the normal heart was present in 11 hearts (73%) of the "right angle" group, in only one heart (8%) of the "acute angle" group, and in none of the "parallel'" group. In contrast, the most common pattern in the setting of complete transposition was observed in none, 8%, and 91% of each group, respectively. Other diverse patterns were recognized in the hearts in the acute angle group, and the incidence of abnormal branching was significantly higher in this than in the other groups (p < 0.01). Knowledge of these anatomic variations in the course of the coronary arteries, some of which would cause problems at either definitive repair or reoperation, are essential for those seeking to achieve optimal surgical repair. (Ann Thorac Surg 1995;59:591-7) here have been numerous techniques described for T anatomic biventricular repair of a double-outlet right ventricle with a subpulmonary ventricular septal defect [1-9], but the optimal one remains undecided. In part this is because this combination of abnormalities includes hearts with various three-dimensional relationships between the pulmonary, tricuspid, and aortic valves, and the ventricular septal defect [3, 4, 10, 11]. Although all examples can be corrected by the arterial switch procedure [5], we prefer to use intraventricular rerouting for those hearts with a side-by-side relationship of the great arteries and, if possible, also for those with an oblique arrangement of the arterial trunks [12]. Our surgical experiences have emphasized, nonetheless, that the origin and courses of the coronary arteries are as crucial as the intracardiac morphologic characteristics when it comes to choosing the most appropriate surgical procedure. In this respect, relatively few hearts have been investigated for the purpose of demonstrating these features in the setting of a double-outlet right ventricle with a subpulmonary ventricular septal defect [4, 5, 10, 11, 13]. In this study, therefore, we analyzed the surgical anatomic features of the coronary arteries in a large series of hearts with this particular anatomic arrangement. Accepted for publication Oct 28, Address reprint requests to Dr Uemura, Department of Paediatrics, National Heart & Lung Institute, Dovehouse St, London SW3 6LY, United Kingdom. Material and Methods Morphologic Study We examined 38 hearts with a double-outlet right ventricle with a subpulmonary ventricular septal defect: 20 by means of preoperative angiography and inspection during surgical repair, and the other 18 through postmortem study. All hearts exhibited the following morphologic features: the usual atrial arrangement, concordant atrioventricular connections, and righthand ventricular topology. Hearts with a severely hypoplastic left or right ventricle were not included. Straddling of the tension apparatus of the mitral valve was seen in two hearts. A coexisting coarctation or interruption of the aortic arch was seen in 15, moderate or severe subaortic stenosis in 19, and moderate subpulmonary stenosis in three. The diagnosis of a double-outlet right ventricle was made on the basis of the ventriculoarterial connection, using the "'50% rule" if the orifice of the pulmonary valve overrode the crest of the ventricular septum [14, 15]. The morphology of ventricular outlets was assessed by determining the angle formed between the plane of the outlet septum versus that of the muscular ventricular septum. This permitted three groups of hearts to be recognized according to the angle between the septums: the group in which an approximately right angle was formed (more than 70 degrees), that in which the planes were parallel (less than 20 degrees), and that in which an acute angle was formed by the planes (between 20 and 70 degrees) (Figs 1, 2). The variations in the aortic origin and epicardial course of the coronary arteries were then compared among the three groups, and also assessed within each group by The Society of Thoracic Surgeons /95]$ (94)01006-X

2 592 UEMURA ET AL Ann Thorac Surg CORONARY ARTERY IN DORV WITH SUBPULMONARY VSD 1995;59:591-7 ~orientation of the utlet septum val~alve pulmonary right coronary artery (RCA) V ~ M A valve J trlcuapid~ ' " ~ / P. valve right-angle r~.. ~~ group _~.d{~ ~i-",vav. ~ I \~ ~:~/f T valve ~ ~ ~ valve parallel group acute-angle group Fig 1. The three patterns of the outlet septum that occur in the double-outlet right ventricle with a subpulmonary ventricular septal defect (VSD). (A valve = aortic valve; P valve = pulmonary valve.) Nomenclature The major coronary arteries were described as anterior interventricular, left circumflex, and right (Fig 3) [16]. In all leftctrcumf~x~\ ~v';~// ~interventricula, artery (LCX) ~ -- J septum in the normal heart anterior ~ } left ~ImmP right posterior the commonest pattern in complete transposition with intact ventricular septum Fig 3. Orifices and courses of the coronary arteries as viewed from above. instances, the arteries arose from one or the other, or both, of the two sinuses adjacent to ("facing") the pulmonary trunk. These sinuses have been termed sinus 1 and sinus 2 [17]. Because some have problems remembering which sinus these numbers refer to, it has also been suggested that the sinuses can be distinguished A B C Fig 2. Three patterns of outlet septal orientation in heart specimens. (A) Right angle. (B) Parallel. (C) Acute angle. (Ao = aorta; PT = pulmonary trunk; arrows = orientation of the outlet septum.)

3 Ann Thorac Surg UEMURA ET AL ;59:591-7 CORONARY ARTERY IN DORV WITH SUBPULMONARY VSD I ~~~~ ~j.~i~ ) #~ Left hand ~'~,~ / ~ ~/s~n;l~i s / Le '\l co ro n a r Y ////Right coronary J I ~ artery / Right~; #2 \, ~.."~"]i ]i ~. coronary ~lartery " j/~_~~ I Le:lhuanO /~--~/~ -Left coronary ~ - ~ ~rtery I A Ca ~ter ny~ i Left coronary artery 4, B ~! -...~.~.~'~eft coronary ~./// I'1 Ig:i~nTn a ~ artery D /\NRig.t coronary ~-~ artery Fig 4. Nomenclature for the aortic facing sinuses. In this study, we used the alternatively proposed description of the facing sinus, which involved the observer looking toward the aorta from the nonfacing sinus of the pulmonary trunk. according to the stance of the observer in the nonfacing aortic sinus looking toward the pulmonary trunk. One sinus is then to the observer's right (sinus 1) and the other is to the left (sinus 2) (Fig 4A). This convention has also produced confusion, however, because in the commonest patterns of the coronary arteries, it is the righthanded sinus that gives rise to the left coronary artery in complete transposition, which, when the arterial trunks are frontally oriented or oblique, is additionally left-sided (Fig 4B). A better way of coping with this problem, suggested initially by Drs Roger Mee and Richard Jonas at the World Forum Symposium held in Hong Kong in November 1992, and proposed independently by Dr Joseph Amato at the Rome symposium of 1991 [18], is to consider the aortic sinuses from the stance of the observer positioned in the nonfacing sinus of the pulmonary trunk. After an arterial switch procedure, of course, the trunk will become the new aorta. Sinus 1 is then the lefthand facing sinus; it is usually left-sided (except with side-by-side trunks, when it is anterior); and, in most patients, it gives rise to the left coronary artery. Sinus 2 is similarly to the right; it is right-sided; and, in most instances, it gives rise to the right coronary artery (Fig 4C). As will be seen, however, there is marked variability in coronary arterial origin from these facing sinuses in the hearts presently described. In this study, we applied the newly proposed description of the facing sinuses as seen from the pulmonary nonfacing sinus looking toward the aorta [18], in case an arterial switch procedure might be one of the surgical choices (Fig 4C, 4D). Results Right-Angle Group All 15 hearts in the group in which the angle formed by the outlet septum and the remainder of the muscular ventricular septum was a right angle exhibited a side-byside relationship of the great arteries, with the aorta positioned rightward relative to the pulmonary trunk. The origins from the facing sinuses and the patterns of courses of the coronary arteries are shown in Figure 5. The most common pattern, seen in 11 hearts (73%), is comparable to that found in the normally structured heart (see Fig 3). In the other four hearts, all three major branches arose from one main stem (single coronary artery). When the orifice of the solitary artery was located within the righthand sinus (sinus 2), an additional artery arose from the other facing sinus (sinus 1) that supplied the musculature of the right ventricular outflow tract as infundibular (conal) branches [16]. In its proximal course,

4 594 UEMURA ET AL Ann Thorac Surg CORONARY ARTERY IN DORV WITH SUBPULMONARY VSD 1995;59:591-7 right-angle group infundibular outlet branch,, (8).... /.septum / left- hand [#11 Lcx interventricular septum 3.ea.s] r l.ea. 11 heartsl ( ) : number of hearts with infundibular branch Fig 5. Orifices and courses of the coronary arteries in the 15 hearts with a right angle between the septal structures. (AIV = anterior interventricular artery; LCX = left circumflex artery; RCA = right coronary artery.) the right coronary artery crossed the surface of the subaortic ventricular mass anteriorly in 12 of the 15 hearts (80%). The main stem leading to the anterior interventricular and the left circumflex arteries traveled posterior to the pulmonary trunk in all but one heart (93%). Blood flow to each branch in the exceptional heart traversed anterior to either the aortic or the pulmonary roots before reaching the area of perfusion. Parallel Group All 11 hearts in the parallel group had the aorta positioned anterior and slightly rightward to the pulmonary trunk. The commonest pattern, seen in 10 hearts (91%), is comparable to that found in complete transposition (compare Figures 3 and 6) [19, 20]. In the remaining heart, a solitary artery arising from the righthand sinus (sinus 2) gave rise to the anterior interventricular, the left circumflex, and the right coronary arteries; a small infundibular artery was the only branch from the lefthand sinus (sinus 1). These infundibular arteries were major branches and took an anteroaortic course in all hearts. In contrast, the main stem of the anterior interventricular and the left circumflex arteries crossed the anterior and leftward portion of the subpulmonary outflow tract in all hearts but one (91%). Acute-Angle Group The relationship of the great arteries in the acute angle group was side-by-side in two hearts, anteroposterior in three, and oblique in the remaining seven. Various patterns of coronary arterial origin were also recognized, including one heart in which the anterior interventricular artery took an intramural course and four that had dual arterial orifices within one sinus (Fig 7). The incidence of branching patterns other than the one in which a main stem giving rise to both the anterior interventricular artery and the left circumflex artery arose from one sinus and the right coronary artery took origin independently from the other facing sinus was significantly greater in this than in the other groups (p < 0.01 by the chi-square test). An additional and independent orifice supplying a small infundibular artery was seen in three of four hearts in the setting of the so-called single coronary artery. The fight coronary artery traveled anterior to the aortic root in three of 12 hearts (25%); this pattern would likely restrict surgical incisions in the subaortic region. A retroaortic course of the right coronary artery was found in the remaining nine hearts. The main stem of the anterior interventricular and the left circumflex arteries crossed the anterior wall of the subpulmonary outflow tract in two hearts, and the anterior interventricular artery took this course independently in five hearts. The anteropulmonary course of the major branches was therefore observed in 58% of the hearts in this group. Comment The morphology of the outflow tracts found in hearts with a double-outlet fight ventricle with a subpulmonary parallel group left-hand (#11,%.(3) I I ~ ~ _ right-hand (3) ~ [#21 infundibular branch AIV LCX CA 10 hearts (1) "~ heart! Fig 6. Orifices and courses of the coronary arteries in the 11 hearts with a parallel arrangement of the septal structures. (AW = anterior interventricular artery; LCX = left circumflex artery; RCA = right coronary artery.)

5 Ann Thorac Surg UEMURA ET AL ;59:591-7 CORONARY ARTERY IN DORV WITH SUBPULMONARY VSD infundlbular / left.hand [#1] AIV~ acute-angle group I LI heart ~~ 1 heart ~eart ~earts ~1 heart ~1 h-~a~ Fig 7. Orifices and courses of the coronary arteries in the 12 hearts with an acute angle between the septal structures. (AIV = anterior interventricular artery; LCX = left circumflex artery; RCA = right coronary artery.) ventricular septal defect (the Taussig-Bing malformation) is markedly heterogeneous. It is precisely these features that determine what the optimal definitive surgical repair should be [3, 4,10,11]. Two particular features are crucial: the relationships of the arterial valves to each other and the orientation of the outlet septum [21]. The clinical importance of these features is then all the greater because they are reported to possibly influence the anatomic arrangement of the coronary arteries [22, 23]. The coronary arterial anatomic characteristics are therefore as crucial as the intracardiac morphologic features when surgeons are seeking the optimal surgical approach, as some of the unusual patterns of the coronary arteries are likely to rule out usually feasible surgical options. When we consider the morphologic features of the ventricular outflow tracts as factors influencing the coronary arterial origins and courses, a suitable method of description and analysis is obviously crucial. In normally structured hearts, the outlet septum is virtually indistinguishable from the rest of the muscular septum, with the aorta originating exclusively from the left ventricle and the pulmonary trunk arising from a sleeve of freestanding infundibular musculature in the right ventricle. Because of this, the aortic valve is positioned posterior and to the right of the pulmonary valve. In keeping with this aortic position, each major coronary arterial branch runs a course that is relatively short and straight (see Fig 3). In the setting of complete transposition with an intact ventricular septum, although the aortic trunk is often situated anterior and slightly to the right of the pulmonary root, concomitant with the discordant ventriculoarterial connections, the outlet septum is again indistinguishable from the rest of the muscular septum and the course of the major coronary arteries is still short in most hearts. The right coronary arteries arise from the righthand sinus (sinus 2), as perceived from the pulmonary trunk, and the anterior interventricular and left circumflex arteries arise from the lefthand sinus (sinus 1) (see Fig 3). In other malformed hearts with abnormal ventriculoarterial connections and less typical arterial relationships, the origins from one or both of the facing sinuses of the aorta are less likely to cause the coronary arteries to take such short and straight courses to the atrioventricular and interventricular grooves. In contrast, long and winding courses are much more common (see Figure 7, for example). In other words, the origin and proximal course of the coronary arteries are likely influenced by how close the facing sinuses of the aorta are to either the atrioventricular or the interventricular groove. Each heart, however, has its own structural features and size, so that, even if a discrete value could be calculated for this distance, and even if it could be acceptably measured, it would be difficult to standardize this informa- tion and to compare hearts. To overcome this problem, and to describe the essential relationship between the facing sinuses and the grooves, we chose to emphasize the angle between the plane of the outlet septum and the remainder of the muscular ventricular septum, in addition to noting the relationship of the arterial trunks themselves, as is traditionally done when distinguishing subsets of hearts with abnormal ventriculoarterial connections [10, 11, 15]. The orientation of the outlet septum was almost always concordant with the plane dividing the arterial valvar orifices. The reason why the arrangement of the arterial trunks does not necessarily represent the three groups employed in this study is due to the fact that the muscular ventricular septum can occasionally assume unusual orientations, producing variations in the location of the interventricular grooves. Such hearts might be described as being rotated in a clockwise or counterclockwise fashion. The definition and clinical application of the angle between the septums warrants discussion. The muscular ventricular septum itself rarely occupies a single fiat plane; it is almost always curved to some extent. For this reason, we defined the plane of the muscular ventricular septum as it formed the floor of the subpulmonary ventricular septal defect. This means that the angle of the outlet septum relative to this part of the muscular septum represents not only the proximity of the facing sinuses to the coronary arterial courses, but also the quantitative malalignment of the outlet septum within the spectrum of double-outlet right ventricle with a subpulmonary ventricular septal defect [11]. This is also of potential clinical significance, as this angle was readily measured morphologically and can be calculated, we predict, by

6 596 UEMURA ET AL Ann Thorac Surg CORONARY ARTERY IN DORV WITH SUBPULMONARY VSD 1995;59:591-7 echocardiography [24]. Echocardiography is almost certainly now the most useful technique for precisely evaluating the intracardiac anatomic features. This diagnostic method should therefore be able to provide crucial preoperative information about the three-dimensional aspects of the arterial valves, and furnish the means for measuring distances and angles for the purpose of describing spatial relationships. Although we cannot yet be sure whether our chosen angle will have echocardiographic utility in distinguishing among three groups of hearts, we are sure that the angle, when defined, will be helpful for surgeons in choosing the most appropriate surgical procedures and, at the same time, in highlighting the potential diversity of coronary arterial origins and courses. Of course, the morphology of the ventricles themselves, in terms of the "balanced" or "unbalanced" nature of their respective size, may also influence the coronary arterial anatomic characteristics. In this respect, no hearts with a small ventricle were included in this study. In the fight-angle group of hearts, the posterior portion of the outlet septum usually meets the posterior limb of the septomarginal trabeculation, together with the ventriculoinfundibular fold. Because of this, the distance between the pulmonary and tricuspid valvar rings is sufficiently large to accommodate construction of an intraventricular pathway from the left ventricle to the aortic orifice [3, 12]. The procedure often requires incision of the fight ventricular outflow tract to prevent an obstruction from forming between the right ventricle and the pulmonary trunk postoperatively [12]. The fact that a major coronary artery taking a course across the subpulmonary right ventricular outflow tract was found in only one heart (7%) clearly supports use of the intraventricular approach in these patients (see Fig 5). In 12 of these patients whose right coronary artery arose from sinus 1 (the lefthand sinus as perceived from the pulmonary trunk), however, the artery crossed over that part of the parietal infundibulum supporting the aortic root. This finding could become crucial if it proved necessary to attempt a right ventriculotomy to release "neopulmonary" stenosis should an arterial switch operation be the option chosen for such a patient (Fig 8). Because epicardial courses of the coronary arteries occasionally cause greater difficulty, due to adhesions, at the time of such a reoperation, rather than at the initial repair, it is important to remember the potential anterior course of the right coronary artery. Indeed, a potentially hazardous course of any of the major coronary arteries should be carefully excluded before any operation is undertaken, so as to ensure that the best individual surgical treatment is rendered. In the hearts with a parallel arrangement of the outlet septum to the muscular ventricular septum, the outlet septum met neither the anterior nor the posterior limb of the septomarginal trabeculation. The three-dimensional features of the ventricular outlets indicate that construction of a pathway from the left ventricle to the pulmonary valvar orifice, with simultaneous performance of the arterial switch procedure, is almost certainly the optimal definitive repair for this group of patients [3, 4, 12]. The RCA the commonest patterns of the coronary arteries in right-angle group & parallel group after arterial switch procedure Fig 8. The coronary arterial courses after translocation of their oririces. (AIV = anterior interventricular artery; LCX = left circumflex artery; RCA = right coronary artery.) observed similarity of the coronary arterial patterns to those found in the setting of complete transposition further endorses this conclusion. In the acute-angle group of hearts, the outer septum is likely to insert directly onto the ventriculoinfundibular fold. The connection between the posterior limb of the septomarginal trabeculation and the outlet septum is either absent or excessively small. In other words, some hearts showed only a slender muscular bridge, and others had no musculature but a fibrous continuity between the pulmonary and the tricuspid valves. The distance between these valvar rings is, in consequence, often too small to accommodate a satisfactory intraventricular pathway from the left ventricle to the aortic orifice (see Fig 1) [3, 12]. Furthermore, part of the tension apparatus of the tricuspid valve is frequently attached to the area where the posterior limb of the septomarginal trabeculation meets the outlet septum, making construction of a tunnel even more difficult. This situation is then complicated still further in that the anteropulmonary course of the major coronary arteries in seven of the 12 hearts (58%) we studied would have been a source of difficulty had the surgeon attempted a right ventriculotomy (see Fig 7). Furthermore, if the arterial switch operation is chosen instead of intraventricular rerouting in these circumstances, the diversity of the coronary arterial anatomic characteristics could again create still more problems. Thus, in our small group of hearts, we encountered an intramural course, dual origins from one sinus, and an additional and independent orifice for an infundibular branch with all three major arteries arising from a single stem within the other facing sinus. However, such coronary arterial anatomic abnormalities, except probably for the intramural courses, are no longer considered a contraindication to the arterial switch proce-

7 Ann Thorac Surg UEMURA ET AL ;59:591-7 CORONARY ARTERY IN DORV WITH SUBPULMONARY VSD dure. The dual orifices within one sinus, nonetheless, might require individual treatment, such as the so-called trap door method, for translocating the coronary arterial button [25]. If one or both orifices are close to the hinge point of the aortic leaflets, which is a frequent finding, this can certainly produce stretching or kinking of the proximal coronary arterial course after translocation. It also needs to be determined whether the additional and independent orifice for the minor infundibular artery should be translocated, and whether the patency of such a small branch, if translocated, would be satisfactory. Should the anatomic arrangement be such as to preclude both arterial switching and intraventricular rerouting, another procedure must be selected. Intraventricular rerouting along with translocation of the pulmonary trunk (the so-called REV procedure), described by Lecompte and colleagues [8], is then an attractive option, because an unobstructed internal pathway from the left ventricle to the aortic orifice could be successfully constructed by resecting the outlet septum and by attaching an intraventricular baffle without using either external conduits or intracardiac grafts. A Rastelli-type operation or the construction of an aortopulmonary anastomosis [2, 3, 7] are also options, but these would then require construction of an external conduit. We conclude from our findings, therefore, that the nature of the coronary arterial anatomic arrangement and its variety is one of the crucial factors that must be taken into consideration when choosing the optimal surgical option for the repair of a double-outlet right ventricle with a subpulmonary ventricular septal defect. Knowledge of the morphologic characteristics of the ventricular outflow tract in a particular patient is important not only for effectively constructing the blood pathway, but also for alerting the surgeon to the likely variety in coronary arterial course. Doctor Uemura is a Visiting Fellow at the National Heart and Lung Institute from the National Cardiovascular Center, Osaka, Japan. Doctors Uemura, Ho, and Anderson's work is supported by the British Heart Foundation. References 1. Kawashima Y, Fujita T, Miyamoto T, Manabe H. Intraventricular rerouting of blood for the correction of Taussig-Bing malformation. J Thorac Cardiovasc Surg 1971;62: Binet JP, Lacour-Gayet F, Conso JF, Dupuis C, Bruniaux J. Complete repair of the Taussig-Bing type of double-outlet right ventricle using the arterial switch operation without coronary translocation: report of one successful case. J Thorac Cardiovasc Surg 1983;85: Sakata R, Lecompte Y, Batisse A, Borromee L, Durandy Y. 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