Chapter 4: Normal and Anomalous Coronary Arteries in Humans. Part 1

Home , Coronary arteries, Coronary artery ectasia, Klaw (character), Myocardial bridge, Pulmonary sequestration

Coronary Anery Anorulies: A Comprehensfue Approach, edited by P. Angelini. Lippincott Williams & Wilkins, Philadelphia O 1999

CHAPTER 4

Normal and Anomalous Coronary Arteries in Humans

Paolo Angelini, Salvador Villason, Albert V. Chan,Jr., andJos6 G. Diez

Part I The presence of a pulmonary circulation organized in series with the systemic circulation had been postulated by HISTORICAL BACKGROUND isolated early researchers: Ibn-na-Nafis, a 13th-century Arab physician working in Damascus; Miguel Serveto, a passion- Interest in coronary anatomy and the nature of the coro- ate 16th-century Spanish theologian; and Cesalpino, a 16th- nary vessels was cautiously aroused in the 16th century, century anatomist from Padua, who coined the term "pulmo- when inquisitive Renaissance scholars began to perform ana- nary circulati on.' 'sz6 Nevertheless, it was not until 1628 that tomic investigations in the early European medical schools. William Harvey (1518-1651), a physician trained in Padua Until then, anatomic knowledge had been heavily influenced but later active in London, and Cambridge, propounded a by the philosophical and theological teachings ofthe ancient, clear, complete, organized concept ofthe circulation, thereby rediscovered masters of the Greek and Arabic schools. Aris- founding the discipline of physiologic anatomy. Discovery totle (384-322 BC), the philosophical interpreter of nature, of the systemic capillary network awaited the introduction and Galen of Pergamum (129-199 AD), the great physician, of the microscope. It was Marcello Malpighi (1628-1694), were the main authorities whose theories continued to domi- operating mainly in Bologna, who first described the circula- nate the medical schools of Salerno, Bologna, Padua, and tion of blood through the peripheral capillary network.s26 eventually Louvain, Paris, and London during the Renais- Regarding the coronary arteries in particular, the founder sance. of descriptive anatomy, the great Flemish anatomist Andreas Leonardo da Vinci (1452-1519), a lone, ingenious spirit, Vesalius (1514-1564), produced a series offundamental /c- examined a few animal hearts (probably of oxen) and also bulae anatomicae (Yenice, 1538), that were followed by his briefly touched on coronary anatomy while exploring the comprehensive treatise "De Humani Corporis Fabrica Libri arcane viscera of the chest.531 His main interest seemed to Septem" (Basel, 1543), which became the basic textbook be in applying the principles of hydraulic physics to cardio- of anatomy for generations of physicians throughout Europe. vascular function. Leonardo tended to rely on instinctive Interestingly, one famous tabula anatomica showed the right curiosity rather than organized, formal methods. He left us coronary artery GCA) originating from the left coronary only brief notes, accompanied by precise, faithful sketches artery (LCA) and coursing anterior to the pulmonary outflow of the coronary anatomy, including the aortic trifoliate valve, tract (Fig.4.2). Similarly, a single coronary ostium was men- the right and left coronary ostia, and the proximal course of tioned by Fallopius (Venice, 1562).so8 Not until 1761, did the right and left coronary arteries (Fig. a.l). He noted that G. P. Morgagni accurately and definitively describe the two the coronary arteries become progressively smaller as they main coronary vessels.so8 During the ensuing centuries, var* progress toward the cardiac apex.s3l He also accurately de- ious investigators published occasional descriptions of pe- scribed the coronary veins and the coronary sinus; his obser- culiar or unusual coronary anatomic features: the work of vation of the arrangement of these structures supported his A. C. Thebesius and R. Vieussens was especially note- assumption that an artery is always accompanied by a worthy.508 vein.s3l Leonardo's approach exemplified the new method With the advent of the 20th century, physicians became of critical, direct observation, which a little more than a increasingly aware of the complexity and variability of the century later would allow better-trained, more-disciplined coronary anatomy. In 1926, this concept was reinforced and scientists to understand how the circulation works.53l put into a biologic prospective by Grant and Regnier,ra6 who qn 28 / Crraprnn 4

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into two branches, one of which (transverse) continues on- ical solutions to be temporarily adopted by common agree- groove ward in the between the left auricle and ventricle. . . . ment. The other (descending) courses along the posterior interventricular furrow. (Gray's Anatomy, 1901 editionsoe) METHODS FOR STUDYING CORONARY Since the beginning of this century, when the preceding MORPHOLOGY passage was published in a leading textbook of human anatomy, there has been a continuously expanding awareness of Traditional descriptions of coronary morphology are the great variability of the coronary anatomy and the diffi- based on anatomic observations in necropsy specimens. The culty of defining normal coronary arteries. This expanding primary tools are gross inspection and fine dissection with awareness has mainly been the result of the introduction of the aid of magnifying loupes. Injection/corrosion methods selective coronary angiography, coronary bypass surgery, involve both injection of a semisolid gelatin mass or of and catheter-based angioplasty. A growing number of coro- chemical fibers (which tend to solidify in response to nary features have become clinically relevant, and an in- changes in temperature) and corrosion induced by changes creasing number of variants have become apparent. Today, in the chemical environment. Injection of radiopaque media, discussions of this subject are far more detailed than would followed by radiography, is also a well-established method have been considered relevantjust a few decades ago. More- for evaluating autopsy specimens. Each of these observa- over, as further developments occur, discussions can be ex- tional methodologies has its indications, advantages, and pected to become even more complex in the future. limitations. So far, the rich literature on coronary anomalies has been marred by a recurrent, inconclusive debate about the defini- Gross Anatomic Inspection tion of coronary normality: What is normal (or unusual but normal) as opposed to atypical, abnormal, abenant,23a'23s' Gross anatomic inspection-obviously the simplest, most s05 I e 1 a anomalous, acces sory, ectopic, incidental, a v ariant,a readily available method for examining autopsy speci- or a less common variant? What is a major or minora2'll5 mens-is often quite adequate. It is preferred for studying 12 t anomaly or a clinically2e'a or hemodynamically2o'e7'23 tit- the coronary ostial anatomy, because it is well suited for nificant anomaly? At a time when confusion still governs describing the location of the ostia with respect to aortic root the terminology and concepts related to coronary anomalies, reference structures (the semilunar leaflets, commissures, we would like to propose a method of study and a discipline and sinotubular junction). Gross anatomic inspection (some- that may promote a more rational organization of the subject times with the aid of magnifying lenses) is also preferred matter. In this method, coronary anomalies are defined by for describing the intrinsic anatomic features of the proximal exclusion, on the basis of a description of the normal coro- coronary anatomy (for example, slitlike ostial ridges in coronary anatomic features. This fundamental organizational nary arteries that originate tangentially with respect to the concept, which one of us (PA) proposed in 1989,480 seems aortic wall). For evaluating a coronary artery's course, distal to have been widely validated since that 1i-".82'tos'3oz gnlt distribution, and termination (as in cases of small anomalous by means of a feature-by-feature description of the normal fistulous communications), the gross anatomic approach is coronary anatomy can coronary anomalies be defined. Some less appropriate: it is not as precise as injection-corrosion features, such as the presence or absence of a common main or radiographic methods. trunk of the left coronary artery @CA), are dichotomous; The great value of gross anatomic inspection lies in its in these cases, normal is easily defined ("normal is to have convenience and negligible cost. Its most obvious limitation a common trunk"). Other features are better described on is its reliance on necropsy material. Indeed, because of the the basis of a continuous spectrum of quantitative data, widespread clinical introduction of precise diagnostic imag- which can be assessed with a normal, or Gaussian, distribu- ing methods (such as computerized axial tomography, nu- tion curve observed in large populations. In such cases, clear magnetic imaging, echocardiography, and angiogra- "normal" should probably be defined as the interval be- phy), necropsy studies are currently performed with tween two standard deviations from the mean value, as com- decreased frequency. monly used in biologic studies. Additionally, the terminology used to distinguish normal and abnormal coronary Injection-Corrosion Techniques arteries should be based solely on morphologic grounds and should avoid the issue ofclinical relevance. Clinical or func- Injection-corrosion techniques are quite satisfactory for tional repercussions of coronary anomalies are obviously showing coronary distribution patterns.le2'3sl'so8 At their important, and they will be discussed at the end of this chap- most sophisticated level of execution, these techniques can ter (see Pathophysiologic Mechanisms and Clinical Implica- allow visualization of even the finest collateral network. tions of Coronary Anomalies). Unfortunately, some ques- However, because this approach depends on comosion tech- tions cannot yet be resolved on the basis of currently niques to better delineate the coronary luminal spaces, it is available knowledge; in these cases, one can only propose inadequate for determining the relationship between coro- certain criteria to be tested in further studies or certain empir- nary arteries and their adjacent structures (such as the depen- Nom,rer eNl ANovrerous ConoNapy Anrnrurs n HuuaNs / 31 dent myocardial segments). Moreover, injection-corrosion ination of a coronary artery from an anomalous aorlic loca- techniques are delicate, time-consuming, and expensive, re- tion or from the anomalous course of a proximal coronary quiring special technical knowledge on the part of the inves- artery can also be reliably identified with echocardiogra- tigator. In recent decades, these techniques have largely been phy,otu especially using the transesophageal approach.aae' 482 replaced by radiologic methods. OL the contrary, the distal coronary anatomy cannot be adequately visualized with these methods.sle's21 related to myocardial perfusion, especially Radiologic Visualization Questions those aimed at ruling out myocardial ischemia in the pres- Radiology may be performed in vivo, as well as in cadav- ence ofcoronary anomalies, can best be resolved by nuclear eric specimens. In recent decades, in vivo selective coronary myocardial perfusion scintigraphy coupled with exercise or angiography has become an unparalleled tool for studying pharmacologic stress testing or by metabolic radioactive coronary anatomy. 2'48' 1zz' 146 3 s s,37 0,442 Because of this meth- tracers coupled with positron emission tomography. In the od's safety, its capacity for precise stereoscopic imaging near future, these testing modalities will become more rele- (achieved by combining multiple simultaneous or sequential vant: they will be used to definitively and categorically es- projections), and its excellent rendition ofcoronary anatomic tablish the clinical relevance of certain coronary anomalies details in motion (by means of enhanced radiologic tech- that are still unclear and to guide the management of individ- niques, digital enhancement, and electronic magnification), ual patients, based on objective evidence of reversible seg- it is currently used in some 2 million patients per year world- mental ischemia. wide. Moreover, it has been used in the great majority of recent investigational studies of coronary anatomy. NORMAL CORONARY ARTERIES IN HUMANS: In cadavers, radiologic single-plane coronary angiography DESCRIPTIONS AND DEFINITIONS inffoduces a serious artifact, related to superimposition of the different planes (the cardiac free walls, septa, atria, and A coronary artery is dejined as any artery or arterial ventricles). This artifact tends to negate the advantage of brqnch that carries blood to cardiac parenchyma (i.e., any greater detail during visualization ofthe fine coronary anat- structure located within the pericardial cavity). The cardiac omy in a nonbeating heart. To overcome this technical prob- parenchyma includes not only the myocardium but also the lem, Schlesinger35l proposed an "unrolling technique" by semilunar and atrioventricular valves, the great vessels (the which the ventricular septum and both atrial walls are elimi- proximal aorta, the pulmonary trunk, and a short segment nated, transforming the cadaver heart into a flat surface. Ob- of the superior vena cava), and the visceral pericardium or viously, this technique introduces other artifacts and pre- epicardium. The parietal pericardium should not be included, cludes examination of certain relevant cardiac structures. so the pericardial arteries should not be considered coronary. Nevertheless, it is an expedient means of studying, for exam- The name and nature of a coronary artery or branch is ple, coronary dominance. defined by that vessel's distal vascularization territory, not by its origin. A coronary afiery that arises from the right anterior sinus of Valsalva and that branches into the left Newer Imaging Techniques anterior descending (LAD) and circumflex territories is not Because of its high cost, coronary angiography is not ap- a right coronary artery (RCA) but, rather, a left main trunk propriate as a primary screening test for ruling out corona"ry with an ectopic origin. Similarly, the dffirent sinuses of anomalies. For this purpose, noninvasive clinical imaging Valsalva are identified not by the coronary arteries that orig- techniques are safer, more.convenient for the patient, and inate from them but, rather, by their own topographic loca- more cost-efficient-particularly echocardiography6'l 23'1 3 1' tion. 33r but also nuclear magnetic resonance imagingee'lo7'222'41'2' When considering the spectrum of coronary morpho- 460'462'463 and computerized axial tomography at rapid rates logies, "normal" should mean "what is commonly ob- of image acquisition. These methods can document the pres- served,t' 3e4'48o and the terms abnormal or anomalous ence of a coronary anomaly or at least greatly raise the level should be used for any form observed in less than l%o of the of suspicion. For providing a thorough anatomic description, general human population.aso This criterion is proposed as however, they are inferior to coronary angiography, not only the dividing line between (l) normality, which would in- because of their intrinsic physical characteristics but also clude the more frequent variations (normal variants), and because their basic approach is tomographic (whereas the (2) abnormality, which would consist of relatively infrequent coronary arteries do not lie in a single plane). Doppler signal variations ( anomalie s ). interrogation can greatly enhance ultrasonographic imaging In essence, a useful convention is all that, on purely ana- by facilitating vessel identification and providing flow ve- tomic grounds, distinguishes a normal coronary artery pat- locity data. Echocardiography, magnetic resonance imaging, tern from an abnormal variant. Only cenain anomalies (such and computerized axial tomography can frequently allow as a coronary aneurysm) predispose the patient to a morbid diagnosis of the larger coronar:y fistulas and anomalous orig- state, and very few anomalies (such as anomalous origina- ination of a coronary artery from the pulmonary artery. Orig- tion of a coronary artery from the pulmonary artery) consti- 32 / Crrerrnn 4

tute, in themselves, a disease state. Pathophysiologic and of-the pulmonary valve, just anterior to the recess between clinical considerations should be clearly distinguished from, the tricuspid and mitral annuli (Fig. 4.4).The posterior wall and subordinate to, anatomic description. of the aortic root is the anterior wall of the sinus transversus With respect to its basic reference framework, the coro- pericardii, a liquid-filled pericardial space that separates the nary artery anatomy should be related to the aortic sinuses aorta from the right and left atria (Fig. 4.4). at one extreme and the dependent myocardium at the other. The aortic and pulmonary valves have a single adjacent The essential reference for describing the origination of the contact point, which is the consistent remnant of the em- coronary arteries is the aortic root. Anatomically, the aortic bryologic aortopulmonary septum (Fig. a.q. This point is a root consists of three equal-sized507 semilunar leaflets, three useful reference for describing the semilunar cusps and si- intercuspal spaces, and three sinuses ofValsalva, as well as nuses. Indeed, the circumference of each semilunar valve the sinotubularjunction, which separates the aortic root from is normally divided into three equal 120' sectors, and the the ascending aorta (Fig. 4.3). In a normal human heart, the aortopulmonary contact point is easlly and consistently lo- aortic valve is situated posterior to-and slightly to the right catable and helps identify the site of one (joining or adja-

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FIGURE 4.3. A and B. Diagrammatic representation of the aortic root in cross- section (grossly sagittal, view A) and in rectified form (unrolled root, view B), after excision of the semilunar cusps to reveal the implantation line of the cusps (view B, arrows). The aortic root is limited distally by the sinotubular junction. The sinuses of Valsalva are limited distally by the sinotubular junction and proximally by the (r'i:t\" implantation line of the cusps. The bottom section of each cusp is shadowed to indicate the underlying muscular septum (view B, solid circle). The intercuspal triangles or spaces (view B, asterisk), have different wall constituents depending on the specific location. View A: AF : anterior fascicle of the bundle of His; AsAO : ascending aorta; IVS : interventricular septum; LA : left atrial wall; M : section of mitral valve; MS : membranous septum; NC : so-called noncoronary sinus; pp : posterior fascicle of the bundle of His; R : right sinus; STJ : sinotubular junction; TS : transverse septum. View B: ICS : intercuspal space or triangle; IVS : interventricular septum; L : left sinus; MS : membranous septum; MV : anterior leaflet of the mitral valve; NC : so-called noncoronary sinus; R : right sinus; STJ : sinotubular junction; SV - sinus of Valsalva. C. Relationship between the aortic root and the right ventricular cavity. AO : aorta, AP : anterior papillary muscle of the tricuspid valve; AR : anterior right pulmonary cusp; C : crista supra- ventricularis; MB : moderator band; P : posterior pulmonary cusp; R : raphe of the right ventricular outllow tract (a residual sign of the fusion line between the embryologic conal ridges); RA : right atrial anteriorwall; SM : crista septo-margin- alis. Shaded area : membranous ventricular septum. 'rouelsod : lsod 1fuepe fueuoul;nd : ueqrr flerceds1'G'V'3tJ Ueeq eqt go eueld Ieuoroc eql ur y4 lroua;ur : '(sesnuls Jur :epoe : gy 6urce; io luecelpe 'seueld leuoSoquo eql ol lcedser qlra peururlelep sr sdsnc aq1) sasnu;s fueuou;nd (a) roUetsod pue (UV) lq6l.rlouelue eqt Jo uonecol e^rlelor eql 'qceordde sqt uI '(,{reuororuou aql ueql gene; req6rq e le palecol sr snurs fueuoulnd (1uec -elpeuou '1y) gal-oralue eqf 'sesnuls (CN) fueuo.rocuou aql pelpc-os) toualsod tqSu puD 'rouatuD {a7 'nualuo TqBu

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Origination of the Coronary Ostia

Normally (i.e., in more thanl%o of individuals),the human corondry arteries have two or three coronary ostia.a2r Whereas two ostia (the right and left) are typically present, it is also normal to see a separate aortic ostium for a conal or infundibular branch, or third coronary utt"ry.tto which is present in 23 to 5lVo of normal h"r1r.1e1'3sl'so8 Less frequently, separate origination of the LAD and circumflex arteries from the aorta, in the absence of a common left main trunk, produces a third (or fourth) coronary ostium. This condition was studied by several anatomists, and the re- porled frequency of absent left main trunk varied from 0.4 to 8.07o, depending on the criteria used by the different investigators.ea'1e2'3st'3e1'413's08 If the defining criterion is minimal evidence of a common left main trunk, however rudi- mentary, like a common aortic niche, the lower estimates FIGURE 4.7. Cross-sectional view of the right coronary cusp, In contrast, criterion is absence of a clearly are correct. if the showing four examples of variant coronary origination: 1, nor- individualized common trunk, the higher estimates are cor- mal, grossly orthogonal to the aortic wall; 2, uplifted; 3, down- rect. The question of whether an absent left main trunk is ward with a tangential path (in a case of ectopic origination an anomaly or a normal variant will be discussed again later from the ascending aorta); 4, horizontal (in a case of low, in this chapter. ectopic origination). The coronary ostia are normally located in the middle of the right anterior and left anterior sinuses, just above the Gig. a.1). This angle has never been precisely studied in upperfree margin of the semilunar leaflets (in the open posi- a large population, and the normal range has never been tion) and just below the sinotubular junction. This general established, but it is important that distinctly unusual angles rule applies to cases involving two, three, or even four coro- of coronary origination be recognized, especially during se- nary ostia. As already suggested in the literaturs,le'23'227'4tz' 480 lective catheterization in clinical studies. Coronary ostia that a specific, detailed anatomic study needs to be undertaken originate ectopically are consistently associated with acute with the aim of establishing, in a large human population ( ' 'tangential' ' ) arterial origination from the aortic wall, and with normal hearts, the spectrum of coronary ostial distribu- only rarely does acute angulation occur in the context of a tion in both the horizontal plane (oriented along the circum- normal ostial location. ference ofthe aortic annulus) and the vertical plane (oriented In size, the coronary ostia are typically equal to, or larger along the longitudinal axis of the ascending aorta). Only than, the proximal segment of the related coronary artery.3ol after a precise description becomes available can the stan- As the coronary arteries produce side branches and progress dard deviations in the two axes be determined and anomalies downstream, they gradually decrease (but never increase) be exactly defined (Fig. a.6). in diameter. Normally, the proximal segment of a coronary artery The course of the coronary arteries is mostly epicardial, arises at a nearly orthogonal angle the aortic wall from at least in humans, although the proximal l-4D is intramural in 5 to 257o of cases, producing a systolic narrowing or milking effect when observed angiographically (see Intra- mural Coronary Artery). The coronary arteries normally ter- minate in the capillary network via arteriolar segments, which are responsible for most of the coronary tree's hemodynamic resistance (see Overview of Coronary Physiology). Direct coronary artery communications with the cardiac cavities or with veins are considered generically abnormal (see Anomalies of Termination: Coronary Fistulas). Although an ideal "coronary luminal size/dependent myocardial mass ratio" no doubt exists, its normal range has been difficult to define. I 75,20 s,23o,233,217,28 1,3o 1,4o8,428,4ee It is important that this ratio be clarified, however, to better representation of the aortic root and FIGURE 4.6. Schematic elucidate anomalies such as coronary ectasia, coronary aneu- the ascending aorta, showing the conceptual pattern of distri- coronary hypoplasia, or absent coronary artery. The bution of the right (R) and left (L) coronary ostia in the vertical rysm, (for the right) and horizontal (for the left) planes (vp and hp, current open debate about how to define these entities stems respectively). 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9t / sNwlnll NI StrrusJuv AlrvNodoD snorvr loNV oNV 'rwQIoN 36 / Cnaprrn4

A bBo FIGURE 4.8. Horizontal cross section of the heart showing the myocardial segments supplied by the LAD (black), RCA (white), circumflex (triangles), and ramus medianus (squares) in cases of dominant RCA (A) and dominant circumflex (B). The numbered circles around the sections represent the location of individual coronarybranches:1 : LAD; 2 : seconddiagonal;3: firstdiagonal;4: obtusemarginal (case A) or ramus (case B); 5,6 = further obtuse marginal branches; 7 : posterior descending artery; 8 : acute marginal; I : anterior right ventricular branch. The section of the papillary muscles is shown inside the left ventricular cavity.

septum, should be constructed to indicate the respective ter- The other atrial branches have a highly variable, unpredicta- ritories of the RCA, LAD, and circumflex artery fig. 4.8). ble morphology. Individually, they are generally considered For clinical angiographic purposes, we support the policy unimportant for clinical purposes, with the possible excep- of describing the coronary a.rtery patterns and distributions, tion of a branch that follows the crista terminalis (the line including detailed mention of the main secondary branches of separation between the smooth posterior section of the (as small as about 1.5 mm in diameter), according to their right atrium and the highly trabeculated anterior portion). territorial distribution (Fig. 4.8). In most cases (about 907o in humans), the RCA is "dominant," meaning that it gener- Left Coronary Artery ates a posterior descending branch, which provides blood flow mainly to the posteroseptal left ventricular myocar- The LCA originates from the middle portion of the left dium, past the proximal, predominantly conductive section anterior sinus of Valsalva, just above the level of the free of this vessel, which gives off only small branches to the edge ofthe open aortic cusp andjust below the sinotubular free wall of the right ventricle. One of the RCA's terminal junction. The exact site of the left coronary ostium varies. branches, which originates at the crux of the heart, provides Specific studies should be undertaken to identify the value nutrient flow to the atrioventricular node. That terminal of two standard deviations in the distribution curve of the branch may facilitate angiographic location of the interven- left coronary ostial site, as proposed in the case of the RCA tricular septum, as the atrioventricular node is situated in Gig. a.6). The left coronary ostium is usually single, so it the cephalad, posterobasal portion of the interventricular is normal to see a common LCA trunk. In an ad hoc review of septum, just underneath-and in front of-the coronary si- 1950 consecutive selective coronary angiograms (see pages nus's opening into the right atrium (Eustachian valve). 38-43), the authors documented absence of a left main coro- Normally, the RCA does not provide branches that cross nary trunk (with double orifices, and separate origination of the anterior interventricular sulcus into the left ventricular the circumflex and LAD arteries) in less than 17o of cases, territory. A small septal (interventricular) branch may origi- a finding that defines this condition as a coronary anomaly. nate from the proximal RCA or directly from the right ante- A proximal coronary vessel originating from the left ostium rior cusp by means of an independent ostium. is called the left main stem or trunk only if it gives rise to Of the multiple small, highly variable atrial branches that both the LAD and the circumflex artery. In anomalous cases, may arise from the RCA, the sinus node artery is the one in which one of these arteries does not originate from the most commonly recognized. Nevertheless, its origin and LCA, the trunk that arises from the left anterior sinus should course vary widely. In 507o of cases, the sinus node artery be called not the left main but the proximal LAD or circum- arises from the proximal RCA. 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r The circumflex artery runs along the left atrioventricular INCIDENCE OF CORONARY groove, descends beneath the left atrial appendage, and courses downward toward the crux of the heart for a vari- ANOMALIES: ANGIOGRAPHIC able distance. An LCA that has a circumflex artery that ANALYSIS OF 1950 CASES does not reach the crux ofthe heart is customarily called "nondominant." An LCA that has a circumflex artery that The recent literature contains several reports about the reaches the crux and produces the posterior descending incidence of coronary anomalies, either in the general patient branch, along the posterior interventricular groove (Fig. population or in patients with clinical evidence of myocar- 4.8), is called "dominant." In reality, however, "right dial ischemia. These reports involve both angiographic and autopsy and left dominance" is a poor term because, as Jamesle2 series. Some of them specifically concern the incidence of coronary anomalies in young persons,8s'106'2a1 ath- noted, the LCA is larger than the RCA in most normal sl8's3a letes,2sa'sta ro16iars,2as patients recovering from an heafts, even when the RCA provides the posterior de- t 43,424 acute myocardial infarctio 11,37' those suffering sudden scending branch. Current terminology referring to domi- death,3e3'st:'s20 or those studied by angiography for sus- nance should be replaced by nomenclature that describes t a'7 6'7 7'196'197'206'406'43o'437 pected coronary artery diseas e.2 the posterior interventricular septum's blood supply or its Unfortunately, the entry criteria and methodology used by distribution, avoiding any reference to literal "domi- the different investigators are poorly described and variably nance.' ' In addition, it is important to realize that the cir- defined, yielding unreliable figures that are not strictly com- cumflex artery and the RCA are not the only arteries that parable. can participate in vascularizing the posterior third of the A higher incidence of coronary anomalies has been con- interventricular septum; another possible participant is the sistently observed in young victims of sudden death than in LAD, which may not end at the apex but, rather, may turn adults undergoing routine autopsy examination (incidence: around it and then follow the posterior interventricular 4 to l5%o versus about l%o, respectively). The medical com- groove for a variable length. For further discussion ofthis munity still hesitates to accept these differences at face point, see page 58. value, however, because the reporting centers tend to have o It is probably useful to define the "minimal circumflex a particular, well-recognized interest in congenital coronary artery" as that vessel which provides at least one branch anomalies; therefore, the incidence of these rare entities is to the territory of the obtuse margin. In cases in which probably atifactually heightened because of a referral bias. the LCA's territory does not extend to the obtuse margin, In addition, the results depend on whether certain relatively one would expect the circumflex artery to originate ectopi- common entities such as muscular bridges are specifically cally (usually from the RCA), and the LCA would be investigated and counted as coronary anomalies. considered an LAD in the absence of a common left main To help clarify the incidence of coronary anomalies, our trunk. group at the Texas Heart Institute evaluated selective coro- o Atrial branches commonly originate from the circumflex nary angiograms obtained from 1950 consecutive patients artery (especially those branches directed to the left with documented or suspected coronary artery obstructive atrium, but sometimes also those directed at the right disease and otherwise normal hearts. atrium and the sinus node's territory). Such branches less frequently arise from the left main trunk, and they never Patients and Methods arise from the LAD. In some 40Vo of normal hearts. the Materials and Techniques artery to the sinus node originates from the proximal circumflex ortery.le2'sos The atrioventricular node artery may Coronary angiograms performed in 2000 consecutive originate from the circumflex artery, but only if the latter cases between January and May 1989 were retrieved from artery reaches the cardiac crux, usually with a dominant the archives of St. Luke's Episcopal Hospital's cardiac cath- pattern. eterization laboratories and were prospectively reviewed according to the criteria described herein. Fifty angiograms In considering the wide spectrum of epicardial coronary were excluded because they either were nonselective or did artery pattems in normal human hearts, one can arrive at the not adequately show all of the expected coronary vessels. following basic conclusion: hearts, in all any left ventricular The remaining 1950 angiograms were technically satisfac- myocardial segment has essentially the same amount of arte- tory and, therefore, were included in the study. The popula- rial supply (measured by capillary density) as any other seg- tion included adult patients (1369 men and 581 women) with ment, but the proximal arterial vessels can be organized into an average age of 56.7 years (Table 4.1). a great number of alternative patterns. A proper, uniform, In the great majority of cases, multiple projections had widely accepted system of nomenclature must be established been obtained by means of the Judkins technique, using pre- to promote reliable interobserver communication, which is formed right and left coronary catheters. 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coronary angiography, when myocardial blushing is fre- TABLE 4.2. lncidence of coronary anomalies and patterns, quently seen, especially in the left anterior oblique pro- as obserued in a continuous series of 1950 angiograms jection, during angiography of both the LCA and the Variable Number Percentage RCA. Coronary anomalies (total) 110 5.64 6. The obtuse margin of the heart (left ventricle) was iden- Split RCA 24 1.23 tified by recognizing the border of the heart in the left Ectopic RCA (right cusp) 22 1.13 anterior oblique projection (at about 45"; see Figures Ectopic RCA (left cusp) 18 0.92 Fistulas 17 0.87 4.8 and 4.11). The circumflex artery was identified as Absent left main coronary 13 0.67 the vessel that coursed in the left atrioventricular groove, artery crossing the obtuse margin of the heart. The larger lat- Circumflex arising from right 13 o.67 eral wall branches, starting from the obtuse margin and cusp LCA moving posteriorly toward the posterior descending ar- arising from right cusp 3 0.15 Low origination of RCA 2 0.1 tery, were labeled the obtuse marginal (OMl), obtuse I Other anomalies 3 0.27 marginal 2 (OM2), and obtuse marginal 3 (OM3). Coronary dominance patterns Right coronary branches. We used the following nomen- Dominant RCA 1641 89.1 clature for right coronary branches: Dominant LCA (circumflex) 164 8.4 Codominant arteries (RCA, 48 2.5 a. "Infundibular (or conal) (Fig. was branch" 4.I2) circumllex) used to designate branches that serve the anterior free : wall of the right ventricular outflow tract (grossly LCA left coronary artery; RCA : right coronary artery. the 3- to 5-cm segment of myocardial territory below the pulmonary valve). These branches may have direct independent aortic origination. Because infun- tuse margin and sometimes including the posterome- dibular branches that originate separately are usually dial papillary muscle (Fig. a.8). smaller than the tip of the diagnostic catheter, selective catheterization of such independent branches Correlations (Table 4.3 was rarely observed. ) b. "Right ventricular branch" was used to designate After identifying the variant anatomic pattems, we related branches that serve the free wall of the inlet and the them to the following variables: sex, the presence of coro- apical portion of the right ventricle. We tried only nary disease (criterion: )507o obstruction of vessel with to identify the acute marginal branch as the artery a lumen larger than 1.5 mm), the presence of a primary that lies closest to the acute margin of the heart. The cardiomyopathy (myocardial systolic dysfunction in the ab- acute margin of the heart was identified angio- sence ofcoronary disease and/or a clinical history ofmyocar- graphically as the lowest point in the "C" described dial infarction able tojustify contractile dysfunction). Coro- by the RCA in the left and right anterior oblique nary anomalies were classified according to the scheme views (Fig. 4.12). c. "Posterior descending branch" was used to designate the branch that follows the posterior interven- TABLE 4.3. Correlations tricular groove and is angiographically identified by Variable Number Percentage the posterior septal penetrating branches (Fig. a.I2). Compared with the anterior septal penetrating Men with coronary anomalies 66/1 369 4.82 Women with coronary anomalies 44/581 7.6 branches, the posterior ones are shorter, frequently Patients with CAD and coronary 63/1 290 4.96 being similar in length to the penetrating branches anomalies in the free wall of the left ventricle. For this reason, Patients without CAD and 57/1950 8.6 our favored means of identifying the posterior de- coronary anomalies scending branch (and, hence, the dominant artery) Patients with aortic valve 7511950 3.8 anomalies was the myocardial blush phase during coronary an- Patients with aortic valve 20175 26.7 giography in the left anterior oblique projection, es- anomalies and coronary pecially with a caudal tilt, when available. We used anomalies the term "codominant circumflex and RCA" for Patients with cardiomyopathy 96/1 950 4.924 Patients with cardiomyopathy 5/96 5.2^'b cases in which two branches, one from each ofthese and coronary anomalies arteries, coursed posterior descending into the Patients without cardiomyopathy 1 05/1 854 5.7b groove, providing septal perforators (Table 4.2). but with coronary anomalies d. 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TABLE 4.4. Classification of coronary anomalies in (normal) human hearts

A) Anomalies of origination and course c) Cx arising from right anterior sinus, with anomalous 1) Absent left main trunk (split origination of LCA) course: 2) Anomalous location of coronary ostium within aortic 1 ) Posterior atrioventricular groove root or near proper aortic sinus of Valsalva (for each 2) Retroaortic artery): d) LCA arising from right anterior sinus, with anoma- a) High lous course: b) Low 1) Posterior atrioventricular groovea or retrocar- c) Commissural diac 3) Anomalous location of coronary ostium outside normal 2) Retroaortica "coronary" aortic sinuses 3) Between aorta and pulmonary arterya a) Right posterior aortic sinus 4) lntraseptal' b) Ascending aorta 5) Anterior to pulmonary outflowa or precardiac c) Left ventricle 6) Posteroanterior interventricular groove" d) Right ventricle 5) Single coronary artery e) Pulmonary artery Variants: B) Anomalies of intrinsic coronary arterial anatomy 1) LCA arising from posterior facing sinus 1) Congenital ostial stenosis or atresia (LCA, LAD, RCA, 2) Cx arising from posterior facing sinus cx) 3) LAD arising from posterior facing sinus a) Coronary ostial dimple 4) RCA arising from anterior right facing sinus b) Coronary ectasia or aneurysm 5) Ectopic location (outside facing sinuses) of any 2) Absent coronary artery coronary artery from pulmonary artery 3) Coronary hypoplasia . From anterior left sinus 4) lntramural coronary artery (muscular bridge) . From pulmonary trunk 5) Subendocardial coronary course . From pulmonary branch 6) Coronary crossing f) Aortic arch 7) Anomalous origination of posterior descending artery g) lnnominate artery from anterior descending branch or septal penetrating h) Right carotid artery branch i) lnternal mammary artery 8) Absent PD (split RCA) j) Bronchialartery Variants: k) Subclavian artery a) (Proximal + distal) PDs, both arising from RCA l) Descending thoracic aorta 9) Absent LAD (split LAD). Variants: 4) Anomalous origination of coronary ostium from oppo- a) LAD + first large septal branch site, facing "coronary" sinus (which may involve joint b) LAD, double origination or adjacent double ostia). Variants: 10) Ectopic origination of first septal branch a) RCA arising from left anterior sinus, with anoma- C) Anomalies of coronary termination lous course: 1 ) lnadequate arteriolar/capillary ramifications? 1) Posterior atrioventricular groove* or retrocar- 2) Fistulas from RCA, LCA, or infundibular artery to: diac a) Right ventricle 2) Retroaortic" b) Right atrium 3) Between aorta and pulmonary arlerya c) Coronary sinus 4) lntraseptal" d) Superior vena cava 5) Anterior to pulmonary outflowa or precardiac e) Pulmonary artery 6) Posteroanterior interventricular groovea f) Pulmonary vein b) LAD arising from right anterior sinus, with anoma- g) Left atrium lous course: h) Left ventricle 1) Between aorta and pulmonary arlery i) Multiple, right + left ventricles 2) lntraseptal D) Anomalous collateral vessels 3) Anterior to pulmonary outflow or precardiac 4) Posteroanterior interventricular groove

a lf a single, common ostium is present, the pattern is considered to represent "single" coronary artery. Cx : circumflex; LAD : left anterior descending coronary artery; LCA : left coronary artery; PD : posterior descending branch; RCA : right coronary artery.

coronary artery inside the atrial septum. Our proposed classi- course," 11s'287 6""urrse a coronary artery's proximal course fication scheme is based on our own experience as angiogra- can be abnormal only if that artery's origin is abnormal (ex- phers and a thorough review of the pertinent literature. It cept in the case of intramural or subendocardial coronary stresses a meticulous, orderly approach in which each arteries). Rather than favoring alphabetical-numerical list- feature of the normal coronary anatomy is viewed as a crite- ings, we prefer descriptive, clear terminology that identifies rion for potential anomalies. We prefer to combine the tradi- each anomaly. Moreover, we prefer to avoid labeling an tional headings "anomalies of origin" and "anomalies of anomaly with the name of its purported discoverer. 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Anomalous Location of a Coronary Ostium Outside the Normal ttCoronary" Aortic Sinuses Ectopic Coronary Ostium Located at or near the Right Posterior Aortic Sinus (Noncoronary)

The right posterior coronary sinus is commonly termed "noncoronary" because coronary ostia are only rarely, if ever, observed in this rinur.77'2le'28s'13s's26,s27'sze A fundamental condition for including a case in this category is the presence of a normal, trifoliate aortic valve. Some of the patients reported in the literature have had, or could have had, substantial anomalies of the aortic valve, mainly of the bicuspid kind. In most true cases of these anomalies, the ectopic ostium has a commissural location: usually it is the LCA ostium that is located next to the posterior left commis- FIGURE 4.13. A. lntraaortic ultrasound image sure. In itself, this anomaly is benign unless it involves a showing the left main coronary artery (LM, white arrow) originating from tangential origin (as it frequently does), in which case it may the square coronary sinus. B. The artery courses posteriorly carry an increased risk of ostial stenosis and/or enhanced around the left coronary sinus and gives rise to the left anterior spasticity. During coronary angiography, this anomaly descending aftery (LAD, white arrow). The ultrasound trans- causes difficult cannulation as a result ofits unexpected loca- ducer's position (in the left coronary sinus in view A and the tion and its tangential or slitlike nature. When this anomaly right coronary sinus in view B) is indicated by the dark central square with the surrounding bright halo ring-down is suspected after initial unsuccessful attempts to cannulate, of artifact. The imaging field is scaled by 8-mm divisions. LCC : left biplane aortography is recommended. The anterior right coronary cusp; NCC : noncoronary cusp; RCC : right coro- oblique and straight lateral projections are the most contribu- nary cusp; RVOT : right ventricular outflow tract. (From Lo tory ones for establishing posterior origination of the LCA. et al. Anomalous origin of left main coronary artery from the The relationship between the ostium and the aofiic sinuses noncoronary sinus: an intravascular ultrasound observation. should be documented, preferably using the right anterior Cathet Cardiovasc Diagn 1997;42:431. Reprinted with per- mission.) oblique projection with a cranial tilt and selective iniections. For selective catheterization, the operator must be highly skilled and patient.17e,21e,3oo.s2e The Amplatz or Multipur- usually involves pose curved catheters offer the best chance the anterior/left surface ofthe aorta.l80'201. of success. Alter- 234'344'3eo In the rare cases reported natively, the Sones catheter, advanced from the brachial ar- in the literature, the site tery, offers a favorable approach. In addition, intravascular of coronary origination has ranged from just above the sinotubular junction ultrasound has recently been used to identify this anomaly to the origin of the innominate artery, sev- (Fig.4.l3).527 eral centimeters above the aorlic valve. This condition dif- This anomaly should be ruled out when selective cannula- fers from the previously discussed milder anomalies (page tion cannot be achieved with Judkins left-sided catheters 45) in that, here, the ostium is clearly located above the and nonselective angiography shows a longer than usual left sinotubular region of the aortic root; the ectopic coronary main trunk. The anomaly is generally considered benign,le, arteries frequently have slitlike orifices and a tangential 186'411 un6 the literature contains only one case in which proximal course along the aorlic wall, on which they lie, origination of the left main artery from the noncoronary loosely attached to the aortic tissue. Occasionally, the proxi- sinus led to a clinical event, namely the occurrence of a mal coronary segment is intramural, inside the aortic wall, large, fatal anterior myocardial infarction in a l2-year-old and is intussuscepted for 0.5 to 5.0 cm.32s Once the ectopic girl.sze The mechanism of coronary occlusion in this case coronary artery reaches the epicardial surface, it regains a was probably clot formation in the slitlike ostium of the normal location and course. The RCA is the most frequently anomalous vessel. ectopic artery, but the LCA (or, separately, the LAD and In the presence of this anomaly, angioplasty of any LCA's circumflex artery) may also originate ectopically. Although branches would also offer unusual difficulties because of an ectopic proximal artery course is not a pathologic condi- precarious selective cannulation and back-up support. tion in itself, the artery may be predisposed to have a more (See Case Reports 4.8 and 4.9 in the Atlas of Case Re- active atherosclerotic buildup, especially at the ostium, per- ports) haps because ofrheologic factors and unusual shear stress.4oe Angiography of this type of anomaly is frequently chal- lenging and incomplete. The basic initial condition that Ectopic Coronary Ostium Arising Outside the Aortic should alert the angiographer is the absence of a coronary Root, in the Ascending Aorta ostium at the expected site. In such cases, the first step is An ectopic coronary ostium located outside the aortic root, to rule out ostial atresia or occlusion, as suggested by retro- in the ascending aorta, can be present at different levels but grade filling of the distal "missing" artery from the contra- 'u€eq IBrIrJou -r^ Jo IeJelBIIoc lelrue8uoc slueserder (cr1e4e Jo crlouels eq esrareqlo uB ur po^rosqo ueeq Jeêu seq uolllpuoJ sIqJ '(9 ,(uur qcrqm) seueue ,(reuoroc lerprecrde Surluur8uo ,(1pru Jeldur{J ees) unrprBcofru eril ol .{\oU luorlnu qlrl( uollll -Jou pue seceds mlnceqeJuelur JelnJulua^ UeI uee.ttleq uoll 6epuluel -eduroc ur 1q3u eql ol poolq fJeuoJoc ulurp splosnu -eJrunru(uoc 'sesuJ qcns u1or.'urudes J?Inculuel lculul qlIA\ erserlu freuourlnd;o sesec ur elculuel lq8u eW uI sJncoo s€ -rs eseql'JeAeA\oq'elolserp Supnp €er.6zr.r€:slesse,t,(reuoroc Surroqq8reu oql olur e1o1s,{s Suunp urerp .(lleuoqcunJ qclq.{\ 'elculuel Uel pepeolreô er{l roJ ruslueqceru Sunue,r e 8ur 'sprosnurs J€lncrJluel tq8u e1dr11nur qlr.&\ lslxooJ urrydes rel -]tr1qsuoc'Jncco ol puel serletuoue Jenel eql op sar,tt ,esr'Els -nculuel lc€lul uB puu BISere.fteuorupd qclql\ uI uoqlpuoJ -eydod,tq relnrulue^ UeI Jo run-rlceds eql uI{lI^\ 'ulser]e Jo sIS -oue1s erolos;o ecueserd eql ur ,(p6 'slerolulloc geeq plrueSuoc xelduoc B JoJ Je{uousru e .(11ensn sI ..elc lelrue8uoc -lrlue^ lq8u eqt uror; ,ftege .{reuoroc u;o uorleur8ug,, ,(reuoroc-pprosnurs pue Ieuuru Jelncrruel Uel-ouoe se qcns serleuroue peluleJun sepnlcxe uorlrurJep sql 'elclJlue^ uol @ n7dor73 aas) a7c7.uua1 eql ruor; (urnrpruco,{ru eql 01 l'tolJ Surpr.tord) lesse,r f.reu ry&ly aW uotg Sutsrty unltso funuotoS cldolcg -oJoc lueulnu e ;o uorleurSuo se peulJep fllcrrls eq plnoqs uorlrpuoc slqJ '(tl't '3rg) s11npe uI 'eAIuA cluoe cllouets B 1(oleq 'e1cu1ue,r ge1 ',{qder8rlurcs reelonu rolpuu luercrJJnsur flplrueSuoc lsnl Ierruou esr^\reqlo u€ ruoq VJU eql;o uorleut8uo Jo Jo sueotu fq.(llercedse 'elqurlsuoruep eq .,(eu urnrpruco.{tu vsv,as.- .r*suoder rrre; e fpo uooq eleq ereql 'e8pelmoul rno oJ luepuedep eql uo lceJJe rllueqcsl uV 'cll€ruelqord elrnb eq ol pelcedxe sr runrlso crdolce eql Jo uoDuzlJeleqluJ elllcel apr4aaA -es 'sesuc qcns q ',,(reue ,fteuoroc Sutleur8uo ,{yptu.rou 'e1rs {al aql uo"tt 3u1s1ty unltsry ttouotoS cgdo1cg -oddo eqt tuo{ .r\olJ lerelelloc;o ecueserd eql uI 'elclrue^ eql olul 1(ou rllolselp eêq 01 uoes eq elclJlue^ uel IIIA\ (syoday aso3 lo IDUV aLil u! 7I'h ttoday asoS aag) frel-ru snoleurou€ eql Uel egl urorg seleur8Fo l€ql 'fcuerc 'sellBruoue qcns -rJJnsur cruoe ecuosqe eql ur ,(lprcedsg lueorJruSrs ;o ,$nuep1 ,{1emr p,tr fqderSorprecoqce euuno1 'eseeslp eAIt 'freue ,{reuoroc eql go erdolce ,{reurrd uror; ,{1r1ue 1ue;e;;tp u -cnlsqo ,fueuoroc ;o ecueprê lecrulTt Jo ecuosqe eql uI ueê se pepre8er eq ppoqs 'eJoJeJeI{l 'pue snolJec ,sruollelncJlc 'seqeuroue oAIBA cruo€ qJns JoJ ,{rolepueur sr ^(qderSor8ue elrlceles Jo sueetu ^{q uorlenp,re ertrleredoe;d elunbepu '(g reldeq3 osle ees) urdolce ,fieuoroc;o {su peseeJcul ue qll.t\ pelercosse eJe serrcruouu oAI€A cruo€ pltue8uoc esneceg ":1,:"#3'J 'lueurecelder eAIeA cruo€ Suunp e q1 1o uo11er 6,,,nonn"' !6 :"?A#XtHllS:.ff flprcedse 'lensn ueql req8rq e 'lossol crdolce eq1 -erder {lqeqord [;euoue srLl1 'lcerl /v\ol1no lelnc1lua^ Uol 1e^te1 lB ;o Jeln^le^qns eql u.roJl (VCU) ftaue fueuoloc lqbp eq1 1o uotleu uo4cessrp InJerec reue 'lno perJJec oq ppoqs furologoe eq1 -r0uo sno;euoue 6ur^ oqs urerOelp clleureqcs 'tl'? fUngll 'frege snopruoue eql Sur8eurep pro^B oJ 'pelcedsnsun sr ,(leurouu eql Jr suelqord pelcedxeun eleerc ,(eur edfl srql;o seueue ,fueuoroc ',{re8rns c€IpJec Suunq 'elqlseeJun sJolo )ve -qlec Surprn8 re8re1 eqt qll^\ uollezueleqlec eÛcelos ropueJ qcrqzrr ur.'(esrnos IeturxoJd pqueSuBl e pue 'ecrSiuo o>Ilplls B 'uorlcrulsqo crsuulut) seJnleeJ lullso Iensnun eql Jo osnuc -eq 'l1ncrJJrp elrnb eq uec slessel oseql Jo ,(lseldor8uy 'srsouels Iepso lno eIru ol 'lueur8es prurxord ar11 Jo sl\er^ leql osoql t-- lerlue8uq ^\oqs uo srseqdure prceds qlrm 'suorlcelord eldqpur uI lno peLLIer zl eq lsruu fqderSor8ue 'pelslnuuec ,(ya,rrlceyes ueeq seq,ftege q freuoroc snoluruoue eql oJuO 'sreleqlec suDlpnl eullnoJ eql .,(lpuorsecco ^{1uo pue (e,t;nc regoqs sll qll/t\'$el eql .(lprced (seuos -se) syepour zlelduy pue 'esodrndqp141 aql eru esod -rnd srqt roJ sreleqlec lngdleq lsour eql 'tunllso crdolce eql el€Inuuec fle,trlceyes pue ^(;quepr ol sJeloqlec lerceds qlr.t peqord oq pFoqs'lueu8es l€relelorelue eq1,(lercedse'egoe Surpuecse eqt 'elup crqderSogoe eueldrq Jo sISBq eqt uO 'f1e.r.r1celesuou q8noqt ue,re 'lesseir snoletuoue oql ezqensll

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Ectopic Coronary Artery Arising from the Pulmonary clinical manifestations and anatomophysiologic forms are Artery not adequately characterized by the terms "infantile" or "adult" or similar fuzzy descriptors (see Chapter 5) but, As a major coronary anomaly that commonly manifests rather, require a more complex terminology. in the pediatric age group,a3'104 ectopic coronary artery origi- ALCAPA varies greatly with respect to clinical presenta- nating from the pulmonary urtery is more thoroughly dis- tion, prognostic implications, and the optimal choice and cussed in Chapter 5. The present chapter covers general ana- timing of therapy. Its treatment depends on multiple anatomic and nosologic considerations regarding this condition. tomic, functional, and clinical variables, including the fol- Anomalous origination of a coronary artery from the pul- lowing factors: monary artery is defined as that condition in which a coronary artery carrying nutrient flow anatomically arises from r The coronary dominance pattern (the larger the RCA, the the pulmonary main trunk.ae Indeed, a coronary artery is better the results of simple ligation of the ectopic vessel also connected to the main pulmonury artery (or its branches) and the overall prognosis) in some cases of coronary fistulas.26 To further confuse this o Obstruction of the ectopic ostium (the more severe the issue, anomalous origination of a conal branch from the main obstruction, the better the natural prognosis) pulmonary artery may result in a condition that resembles o The extent of acquired coronary artery obstructive disease a coronary fistula both angiographically and anatomically. (the more extensive the disease, the more severe the myo- Interestingly, in anomalous origination of a coronary artery cardial ischemia) from the pulmonary artery, the direction of the fistulous flow o Epicardial versus intramural collateral patterns (the for- is toward the pulmonary artery, 8'113'306'318'33e'452 as seen in mer pattern promotes fistulous flow, and the latter one coronary-to-pulmonary fistulas. Nevertheless, in the present favors nutrient flow) condition, blood flow originates from the contralateral, nor- o The myocardial oxygen demand (which is related to left mally originating, coronary artery; whereas in coronary-to- ventricular dilation, diastolic volume overload secondary pulmonary fistulas, flow originates from the coronary aortic to a left-to-right shunt and mitral regurgitation, and sys- ostium of the artery that has the fistulous communication. temic vasoconstriction) The following forms of ectopic origination of one or more . The pulmonary artery pressure (the higher the pressure, coronary arteries from the pulmonary artery have been re- the lower the fistulous flow)276 3' r97'27 7'27 a'426 o p orted.T Body weight and lifestyle factors

o Anomalous LCA arising from the posterior facing sinus Less common, clinically different conditions-anoma- of the pulmonary afiery or from the pulmonary trunk or lous origination of the RCA, LAD, or circumflex artery from branches (the most common form, abbreviated as AL- the pulmonary artery-are presented here, as they are usu- cAPA)467 ally compatible with prolonged, frequently asymptomatic, o (Isolated) circumflex artery arising from the pulmonary survival in the adult age groupl. artery (posterior facing sinus) or one of its branchessT'74' 169,292 Anomalous Origination of the RCA, I-AD, or Circumflex r (Isolated) LAD arising from the posterior facing sinus of Artery From the Pulmonary Artery the pulmonary urt"rynt Each of these three anomalies has different pathophysio- o RCA originating from the anterior facing sinus of the logic consequences and clinical presentations. Origination pulmonary artery or from the pulmonary trunk or its I of the RCA the pulmonary artery has been described branChes o2,25 1,264 37 a,42o from in several recent isolated case reports.l l7'130'214'226'2st'252' o Simultaneous RCA and LCA arising from the pulmonary 264'312374347'4o0'416'42,J In most instances, the ectopic ostium artery3e8'+sr'+6s'a7a (sometimes originating in a single com- was described as being located at the anterior right pulmo- mon trunkso3) nary cusp, and the RCA was dominant, with a posterior de- o Small (right infundibular or conal) branch arising from scending branch. In such cases, collateral circulation be- the anterior facing sinus of the pulmonary artery tween the LCA and the RCA follows the patterns seen in Occasionally, ostial stenosis involving a ridge or fibrous atherosclerotic occlusion of the coronary arteries: the atrial, buildup is observed in the pulmonary arterial wall.2s2 This infundibular, right anterior, and septal branches may contrib- condition may significantly decrease fistulous flow. ute to variable degrees in individual cases (Fig.4.l5A). The Anomalous origination of the entire LCA from the pulmo- septal branches are usually the dominant source of collateral nary aftery (ALCAPA) has distinct clinical features that are flow (from the LAD to the posterior descending artery). discussed in Chapter 5. In the literature and in clinical prac- These enlarged vessels, with their fistulous flow. are promi- tice, several taxonomic classification criteria have been pro- nently displayed not only during angiography but also during posed, mainly in response to the clinical need for distinguish- echocardiography with Doppler interrogation (Fig. 4.15). In ing between subtypes of ALCAPA that have different adults, the condition is typically recognized because of a prognostic and therapeutic implications. 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/ SNWtng NI Ssrf,trJt{V SnO'IVT\IONV ONV TgInItIO\l 6V ^){VNOrIOD 50 / CHaprsR.4 aorta with the aid of extracorporeal circulation and transec- descending thoracic aorta63 in humans. Most of these reports tion of the pulmonary artery (see Chapter 5). The ectopic mention accompanying major congenital heart defects.63.330 circumflex artery more typically originates from a pulmo- In such cases, the proximal coronary trunk's ectopia repro- nary branch (the proximal right or left), and reimplantation duces, in humans, the normal coronary pattern seen in var- necessitates careful dissection. ious animals (see Chapter 1). Reports of bronchial origina- Unlike in ALCAPA, the ratio of fistulous to nutrient flow tion of a coronary artery (see also Coronary-Bronchial seems to favor nutrient flow, so that myocardial ischemic Fistula, page62) should be examined critically: because both manifestations tend to be more limited and to occur only the bronchial and the coronary arteries are normally subject with maximal exercise. Frequently, a relatively obstructive to the same systemic pressure, no flow could be expected ectopic coronary ostium will also limit the fistulous flow. in the absence of a congenital coronary obstruction or a su- The amount of absolute flow probably perpetuates an intrin- prasystemic pulmonary pressure with an inverted patent duc- sic mechanism of progressive enlargement of the involved tus arteriosus (usually in the presence of a hypoplastic left l03 vessels. heart syndrome). In cases of uncomplicated extracardiac Although ectopic origination of a coronary artery from origination of the coronary arteries from the systemic circu- the pulmonary artery is routinely subjected to surgical repair, lation, no myocardial ischemic effects are generally ex- the need for repair has not been well established.l2s In the pected. absence of major clinical manifestations (recurrent angina, myocardial infarction, venfficular arrhythmias, syncope, or aborted sudden death), anomalous origination from the pul- Anomalous Origination of a Coronary Artery from the monary artery may not, in itself, be an automatic indication Opposite, Facing Sinus of Valsalva for surgery, especially if the anomalous vessel is a smaller As stated earlier, the right, left, LAD, and circumflex coro- one such as a nondominant RCA or a circumflex artery. In nary arteries are defined by virtue of their territory of distri- such cases, stress testing is frequently negative forreversible bution, not by their origination.taT When a coronary artery ischemia in adult patients, although mild fixed myocardial arises anomalously from the opposite-from-normal sinus of uptake defects are frequently found on nuclear images, be- Valsalva, the artery's intrinsic name and nature (or function) cause of old scar tissue and/or a rich collateral network, remain unchanged, and only its origin and proximal course which replaces myocardial tissue. 12s'2@ Progressive enlarge- are anomalous.2o8 Of necessity, the artery's proximal course ment of the dilated coronary vessels, with the risk of intimal is abnormal in these cases, as the artery connects with the changes, mural thrombosis, and/or accelerated atherosclero- contralateral, in situ vascular network. Because these anoma- sis, is a possibility in these cases, just as in primary coronary lies are chNacteized by abnormal origination of an other- fistula. This factor tends to encourage early intervention wise "normal" coronary artery from the opposite sinus of (during childhood or the patient's teen years), because other- Valsalva, it is important that the essence of a coronary artery wise the extremely dilated vessels with increased flow would (in contradistinction to, and independently of, its origin)be be transformed, by surgical correction during adult life, into clearly defined. aneurysmatic vessels with normal flow, yielding a persis- Indeed, conceptually the human heart has not two, but tently poor prognosis because of the risk of mural thrombo- three, coronary arteries: the RCA, LAD, and circumflex. The sis. After correction of this anomaly, coronary ectasia may essence previously undergo reversal in young patients but will not generally do of each of these arteries was discussed (pages 35-38), and it becomes clear in light of the spectrum so in older ones. The survival of untreated older patients is possible an indication, if not proof, of the benign nature of the anom- of variations described in this section. aly in such instances. Moreover, the surgical risks may be The RCA is essentially that artery that courses in the right substantially greater and the potential benefits fewer in older atrioventricula"r groove and provides nutritive branches to patients, causing many physicians to prefer continued medi- the free wall of the right ventricle. Branches that supply cal treatment and some surgeons to prefer simple ligation the right ventricular infundibulum, or conus, often originate (versus the more complex reimplantation) of the ectopic directly from the right aortic sinus and are not an essential vessel. part of the RCA. The same is true of the sinus or atrioventric- (See Case Report 4.1 I in the Atlas of Case Reports) ular nodal arteries and the posterior descending branch: all of these branches may originate anomalously without chang- ing the nature of the RCA. Ectopic Coronary Arising the Aortic Arch, Ostium from Similarly, the LAD is essentially that artery that courses Carotid Artery, Internal Innominate Artery, Right along the anterior interventricular groove and provides per- Mammary Artery, Bronchial Artery, Subclavian Artery, forating branches to most of the anterior ventricular septum. or Descending Thoracic Aorta It is not essential that the LAD provide a diagonal branch The literature contains rare reports of extracardiac origina- (although it usually does) or reach the apex, but it is essential tion of the coronary arteries from the aortic arch,63 innomi- for the LAD to course mostly in the anterior subepicardial nate artery,86 right carotid artery,63 internal mammary eir- space. 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FIGURE 4.17. Conceptual diagram showing most of the possible paths (1 through 5) by which the RCA, LAD, and circumflex anery (Cx) can potentially connect with the opposite coronary cusps. Paths: 1, retrocardiac; 2, retroaortic; 3, preaortic, or between the aorta and pulmonary artery; 4, intraseptal (supracristal); 5, prepulmonary (precardiac). The aortic and pulmonary cusps are labeled according to theirpositioninspace: AL:antero-left; AR:antero-right; P:posterior; M:mitral valve; T: tricuspid valve.

path most commonly associated with this type of anomaly, replacement might compromise the aberrant vessel. Other- specifically involving origination of the circumflex artery wise, the anomaly, in itself, is not expected to have any from the right sinus of Valsalva.al'334'36s This anomaly's clinical consequences.336 incidence in the general population ranges from 0.1 to The same retroaortic path can also be observed, although 0.9Eo,2t"'4r including cases in which the circumflex artery less frequently,32a when the RCA originates from the left has a separate origin, adjacent to the RCA ostium, and those sinus (either directly or, more often, from a common mixed in which the circumflex artery arises jointly with the RCA trunk), and when the entire LCA originates from the right from a common short, mixed trunk. The anrmalous retroaor- sinus. The retroaortic path is not seen, however, when the tic circumflex path courses just next to the posterior wall of LAD has an isolated ectopic origin. the aorta, in the sulcus between the atria and the aorta (the Path 3, the preaortic anomalous path, (Fig. 4. 17) courses transverse sinus), and finally reaches a normal location in "between the aorta and pulmonary artery. 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TABLE 4.5. Single Coronary Arteries: Classification creation of an ostial obstruction (by means of a large guiding Criteria catheter or any other instrument, such as the bulky direc- Sinus of Origination tional atherectomy device) would be poorly tolerated and 1) Right anterior sinus could cause symptoms (angina, dyspnea, light-headedness) 2) Left anterior sinus and important changes in physiologic variables, including 3) Posterior sinus 4) Ectopic sinus, at: blood pressure. In general, angioplasty of the common trunk A) Ascending aorta is absolutely contraindicated, even with the use ofstents, not B) Systemic artery only because of the increased perioperative risk but, more C) Pulmonary artery importantly, because of the risk of postoperative restenosis Pathways Followed by Each Ectopic Branch Arising From the resulting in sudden death (a risk that is expected to be even Proximal Trunk' 1) Retrocardiac (circumflex, LCA or RCA) higher than after angioplasty of the left main trunk). 2) Retroaortic (circumflex, LCA, or RCA) During coronary artery bypass surgery, the presence of a 3) Preaortic (LCA or RCA) single coronary artery should not affect any technical deci- 4) lntraseptal (LCA, LAD, or RCA) sions, except fbr suggesting the absolute need for as many 5) Precardiac (LCA, LAD, or RCA) arterial conduits as possible. Proximal mixed trunk obstruc- a Any individual case may involve more than one anoma- tion is particularly relevant in this regard, because it tends lous path. to progress to total occlusion soon after successful bypass surgery. In contrast to venous grafts, arterial grafts are expected to provide longer-lasting conduits. If graft occlusion location, their sequence of origination, and the proximal does eventually occur, it will likely be fatal because of the course of their anomalous coronary branches.362 Excluded presence of total occlusion of the native circulation. from this list are several conditions that resemble single coronary artery but involve ectopic origination of a coronary artery from the opposite cusp, from an additional ostium ANOMALIES OF INTRINSIC CORONARY adjacent to the normal coronary ostium for that cusp. To ARTERIAL ANATOMY conclusively establish the diagnosis of single coronary ar- Congenital Ostial Stenosis or Atresia tery, angiographers and anatomists should verify (1) the l'1 3e'337'423 presence of a single ostium in one sinus, jointly with the The literature contains occasional repofis3o's o1 absence of an ostium in the opposite sinus, and (2) the lack coronary arteries that are u1aa1i"l60'ass'a7s'4e2 or stenosed be- of origination of any other coronary artery from an ectopic cause of a membrane or fibrotic ridgeles located at, or near, site. This process is usually quite simple for anatomists but the aortic orifice in an otherwise normal heart. In cases of may be harder for angiographers, who may encounter diffi- atresia, only a dimple is seen from the aortic side.13 Histolog- culties in ascertaining that the arlery in question indeed sup- ically, when the obstructive element is congenital, it invaria- plies all ofthe heart and that no additional ectopic coronary bly consists of fibrous tissue. A stenosis observed during artery exists. angiography, surgery, or autopsy may be subject to debate Functionally, single coronary aftery has essentially the regarding its nature (congenital versus acquired;.13e'337 The same clinical implications as ectopic coronary origination condition may be associated with a coronary anomaly, some- from the opposite sinus but with separate ostia; nevertheless, times involving tangential origination of a coronary ar- a single coronary ar1ery is not as susceptible to tangential tery.326'z+o Indeed, atherosclerotic growth may occur early origin or ostial ridge pathology as are ectopic coronary a.rter- in life at the site of a congenital ostial fibrotic plaque.2se ies with independent ostia. Coronary blood flow is not af- Coronary ostial or proximal occlusion frequently occurs in fected by the simple presence of a single proximal trunk that the context of pulmonary valve atresia with intact ventricular supplies coronary flow to the entire heart, unless congenital septum (see Chapter 6); in extreme cases, both the RCA or acquired obstructive disease is present in the proximal and the LAD may be affected by ostial atresia.3e6 Isolated mixed trunk.22'157 lnsuch a case, the hemodynamic repercus- coronary ostial atresia (total occlusion) probably represents sions would be quite severe,24'423 as the whole heart could the extreme degree of improper formation of the aortic os- become ischemic without having any possible source of col- tium, and its presence often raises two questions: first, is lateral circulation. Although definitive studies are not avail- the condition congenital or acquired, and second and more able, the incidence of atherosclerotic disease does not appear importantly, is it truly a case of ostial atresia or is it anoma- to be increased in the mixed trunk. An ectopic single coro- lous origination? The congenital cases could be regarded as nary ostiuml80'le8 could, indeed, be more susceptible to con- instances of neonatally or fetally acquired ostial occlusion, genital (ostial) or acquired obstructive disease, but the rarity which occurs after a normal distal coronary tree has already of this anomaly precludes adequate analysis. developed (however, such occlusion would most likely occur During coronary angioplasty, a few minor adjustments are after embryologic development). Ostial atresia in the left necessary in patients with a single coronary artery.21'rae' corona.ry system can potentially occur, not only at the aortic 37s'421 1n the presence of a single ostium, even the temporary connection site but also at the left main bifurcation, the site 66r'lueu'IdoloAeP o1 pedleq eleq sursfrneue ,(reuoroc ur ,{1rco1e,r ,!\oU poolq cruo.{rqrue Suunp slueutnu pnuesse Jo {cel e Jo llnser € se eJnsBeru ol pesn sr eJr,{\ urrog ,(reuoroc relddoq u gJItI1Y\ ur unrprucofur luepuedep eql;o ersuldod,(q ot peel o1 pelcedxe selpqs ',{reue .(reuoroJ pezrs-pruJou esr/y\JeqJo ue ur suorl sr ,(rege f.reuoroc u Jo ecuesqu (cr3o1o,ftqure) lelrue8uoc -BIrp pezrleJol se peurJep e:eryr urs,(rneue pu€ ersBlce 'relerue 'uorlelueruncop eruJ elenbepe Jo ocuesqe e4l ul vzz.strso.vt -lp freue freuoroc Ieuuou Jo uorssnrsrp SuroSuo; eql u1 ,rzsaqJuDJq Jo sauauo {touo.toc ,,3utssttu,, Qtuatnddt f,gt1 usttnauy ro Dlst ttouo.rol -uepr ol posn'Jeurousnu e .,(lpreue8 sr ernlelcueuou srqJ tJg ,fte1ry,(reuo.ro3 luesqy '(7 reldeq3 ees) ;1es1r il€.l.r eql Jo uortedrcrlred ea.rlcu 'luepued -epu eql lnoqll.Â llea cllroe eql olul uotlerleued ecnpul ol aso3 IDUV puD g'2 aag) (syoday lo aql ut t ['t syoday aso3 stuees >IJoA\leu ,(reuoroc e,trlrurrrd eql 1l?ql eJueprAe lueceJ Jo ',(3o1o 1q311 ur .(lercedse 'r€elcun ere suorlecrldun luluerudole,rep -r1e perrnbcu ue eJuaprê 8uoJls peJeprsuos eq uuJ elull Jo sU esr'pne ,(reuo.roc crSolo,trqure ue Jo lueuruer eql eq ,{eu qlr.{\ ruslftnauu ,(JeuoJoc eql Jo uollnloser lenper8 pue sp eldurrp sqt q8noqtly freuoroc € ol psel lou seop 'eseesrp ,nr',fteue -ueue Jo ,fuo1srq pelueunJop ,(Fue1c v eq1;o se3u1s leqt uoruod pgrr sU ur uorsserdep B sur{ ueeq ,(ruuoroc-orvr1 elnce eql ut ,{1uo pcrd,$ ere s8utpug cr3oyo1sr11 (rouelsod 6zz'lefig Ieuuou e ur snurs tq8u) 'ftuuorocuou eqt (7) ro -celuoc eJoru uele sr uorlJurlsrp eql 'sqnpe vI 77s,o67,g7;sll runrlso freuoroc e18urs eql q1,ln euo eql elrsoddo snurs eqt (1) -ueue s,r{eserrru) ur se 'uorleJoue8ep lerperu uorg 3ur1ps Jeqlre qtrqa ur sesec 01 JeJeJ e,1\ re'.rr'suopeclldrur crleueSo -eJ srus.(rneu€ perrnbce ruor; qsrn8urlsrp ol pJeq fpuenber; -,{rqure etuos eleq,{Eru tuql (uouuur8uo ,fteuoroc leuorlcunJ ere surs,{rneue freuoroc ,ftetuud 'e8u culurped Ielrue8uoc ;o pe,,rrrdep snurs cruoe uu Je euoe eql Jo IIeA\ eql ur uors Sluerlsd ur ue^g IBJnur puB uorl JO rLv,rL1',6oz,8lt,te'IqruoJql -serdep u) Surpur; snoletuoue ue roJ pesn ueeq setl ..e1drury -BJecln .,(11eque,,r.e pue'Suruelcrql ptunul'uotlureue8ep lerp 1UrlSO ,ftUUOrOC,, luJel erlJ rer-.rr'eJnlBJAlrl lueCeJ eql uI -e{u qlr,4a'r(ruoluue 11e,t. .,fteuoroc crsulJlul Ieuxouqu,(g8tq e e,req flpnsn erselco frepuoces ro ,fteuud qlrl\ stuerled aldurlg 7m7sg ttouo"roS qlrurr s8'sulnlsrJ IeJerrre J-oJeuoroc re8rul sluerled replo ur pue sI uees f1ecrd.ft sI uollel VdVJ'IV (syoday aso3 lo IDUV ary u! r€'r ltoday asoS aag) -rp pursfrneue,fuuuoroc pezrlecol'eleuonrodordsrp'puo11lp oor,rra'UrnrlSO r(reUO -pV 'Dtsopa Ktowui ur sD pasDanap pu 'pasDanur Q1on1n -roc Surfl.repun eql Jo uorl€Jelrlqo ol speel qJrq.{4. 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As suggested by R. D. Leachman (oral communication, sies (0.00247o), according to the dubious criterion of an "ab- 1989), some cases of syndrome X (angina and myocardial sent dominant vessel" (see Absent Posterior Descending ischemia in the absence of coronary obstruction) may be Branch). caused by a defective number of capillaries per myocardial (See Case Report 4.35 in the Atlas of Case Reports) fiber. This syndrome has never been definitely documented and is not identical with what has been called "absent coro- Intramural Coronary Artery (Muscular Bridge) r'ary artery" in the literature. On angiographic grounds, the most frequent reasonses for On anatomic grounds, the general rule in human hearts an apparently missing coronary artery are coronary ectopia is that large coronary arteries and their branches are situ- subepicardial (misdiagnosed), coronary occlusion with lack of demonstra- ated in the loose connective tissue of the space. Nevertheless, the septal penetrating branches are ble collateral retrograde fi.lling,22s'433 or an alternative coro- normally intramyocardial, and other usually subepicardial nary artery tree pattern that may not be recognized on angi- branches are found to be intramural in more than lVo of ography. Although the literature includes sporadic cases in instances.68'ls7'3 I 6 which an absent coronary artery was reported to cause chest In several mammals and in birds, most of the coronary pain,r78 cardiomyopathy,3a or a myocardial infarction,z2a ab- arteries are intramyocardial (see Chapter 1), apparently with- sence of a coronary artery has never been established as a out having adverse functional consequences. Three consider- specific congenital entity. ations are relevant in defining and discussing muscular (See Report 4.6 Case Reports) Case in the Atlas of bridges in humans: (1) nosologic considerations (what con- stitutes a muscular bridge? is it an anomaly?); (2) functional Coronary Hypoplasia considerations (are muscular bridges able to cause disease?); and (3) prognostic considerations (do muscular bridges lead Several reports have appeared in the literature alluding to to unexpected pathologic events such as spasm, thrombosis, a poorly defined entity called coronary hypoplasia.8T'13'7'26t' or atherosclerotic changes?). 322'a2s Butli"t in this chapter, we discussed the difficulty of An intramural coronary ar.teryl36 is defined as a coronary demonstrating congenital inadequacy of coronary vessel artery that has a segment of variable length covered by myo- size. Normal coronary arterial size should be defined in cardial fibers but that otherwise lies subepicardially.266 terms of both resting metabolic needs and coronary reserve. These fibers constitute the "bridge," whereas the underlying Epicardial coronary branches normally maintain an ideal lu- coronary segment is not the bridge but, rather, is the minal diameter ratio with respect to the dependent myocar- "bridged artery." Fine anatomic dissection, with the use of dial bed or territory or capillary network.2os'23o'233'z8t'3ot microscopy, has indicated a high incidence of myocardial Gouldras theorized that flow velocity is the most practical fibers overriding otherwise subepicardial coronary arteries parameter for measuring the adequacy of vessel diameter: a or branches, as reported in detail by Polacek.306 higher than normal flow velocity would imply a vessel size In clinical angiographic studies, detection of the intramyo- that is restricted in comparison to the distal arteriolar-capil- cardial course of a coronary artery depends on systolic lary network. However, this type of measurement was previ- compression,e'3o7 a narrowing of the lumen ("milking ef- ously quite impractical; it became clinically possible only fect") seen during systolic myocardial contraction. Phasic narrowing of a coronary aftery may also occur in other condi- recently, with the introduction of flow velocity wires, and tions, such as in the presence of ventricular aneurysms or has not yet been used to substantiate the claim of coronary pericardial fibrous bands.lo This angiographic marker is hypoplasia. A more practical diagnostic method may be highly predictive of an intramyocardial coronary course, but based on the simultaneous (1) angiographic appearance of it is actually seen in only a minority of anatomically detecta- a "hypoplastic" coronary branch (that has a small diameter ble cases. Administration of a vasodilator (typically, intra- with respect to the apparent area of dependent myocardium) coronary nitroglycerin in a 100- to 300-/rg bolusr2'187) and (2) demonstration of local reversible ischemia (reduced greatly facilitates the angiographic recognition of systolic reserve) during stress testing with myocardial nu- coronary narrowing. Also, multiple angiographic views of the in- the clear scintigraphy. We are not aware of any cases in volved vessel may add relevant information. Systolic nar- literature in which the diagnosis of hypoplasia could be rowing is generally considered to be caused by coronary soundly based on such combined evidence, and any report compression by myocardial fibers that are oriented circum- that portrays a small coronary artery as a pathologic congeni- ferentially with respect to the heart (and tangentially with tal entity47z should be viewed with skepticism. In most cases, respect to the involved vessel). It is usually best seen in the terminology is used incorrectly, and the dependent myo- projections that are tangential to the cardiac wall over which cardial bed is actually served by alternative sources (unusual the involved artery is located (see Case Report 4.36 in the coronary patterns),322 or coronary spasm or diffuse disease is Atlas of Case Reports). A less reliable, indirect indicator of present. Roberts and coworkers322 observed "hypoplastic" an intramural coronary segment is the "U sign,"12 caused right or circumflex arteries in 8 of 3400 consecutive autop- by the artery's subclinically accentuated descent from its -ce1e u ueql eJoru eq FBru freue .{muo.roJ eIqezIS € Jo uoll€col Suunp uorlcnpord el€lcel Ieco-l zsv,ozv, tsz,srz.noz.eer.zsr Iensnun srqJ 6rr'snlnuuu oAI€A prdsncul eql surolpe (unu1e ,ur.r'se8prrq Jelncsnu Jo uorlceseJ lecrSrns ro 3ut1ue1s .(reu ged pue tq8rr eql;o rezrrol eql ur 'xruc eqt ol lerurxord lsnD lueu -oJoJ Jeue JerleJ pe^Jesqo e^€q sJoqlne eluos 6r€.rzE.s0,

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Lg / sNwtnll NI sarurJdv nrvNodo3 snorvrvoNl\y' qNv ryt{doN 58 / Cruprrn 4 curiosity, especially during surgery for debridging of a coro- Absent Posterior Descending Branch (Split RCA) nary artery, or tricuspid valve replacement or valvuloplasty. As a rule, the posterior descending branch is a single, The literature includes repofts of cases in which a simple, continuous vessel that originates from the RCA or circum- nonextracorporeal approach to correcting a muscular bridge flex artery, at the crux of the heart, and courses in the poste- of the LAD became a surgical nightmare because of perfora- rior interventricular groove. Occasionally, the posterior de- tion of the right ventricle during unroofing of a segment of scending branch comprises two segments: one that originates the intramyocardial LAD in an unexpected subendocardial normally from the distal RCA at the cardiac crux and courses location.eo Unfortunately, no angiographic clues allow this only in the upper posterior portion of the interventricular anomaly to be diagnosed before surgery. A subendocardial groove; and another segment that originates from the mid coronary artery might be viewed as an intermediate stage in RCA, close to the acute margin of the heart, and reaches the a spectrum of "coronary malpositions" ranging from the distal posterior portion of the interventricular groove.147'470 normal subepicardial location to intramyocardial coronary Altematively, the LAD or circumflex artery may supply part artery and to coronaro-cameral fistula. or all of the posterior descending branch, causing it to appear interrupted or split (Fig. 4.18). This phenomenon is a noso- Coronary Crossing logic curiosity, but it may become clinically relevant in surgical grafting of the "posterior descending As a rule, epicardial coronary arteries do not cross one branch" or while attempting myocardial scintigraphic/coronary angiographic another. The literature contains only a few angiographic (but not anatomic) reports2T3 that describe crossing of adjacent correlations. branches, apparently at the subepicardial level. This phe- (See Case Reports 4.38 and 4.39 in the Atlas of Case nomenon should not be confused with superimposition of Reports) coronary branches during angiography when the vessels lie in different planes. In almost all reponed cases and in the ttAbsent LADt' few cases seen by these authors, the crossed arleries were obtuse marginal branches. By examining this feature in sev- In the human heart. the anterior subdivision of the LCA eral angiographic views, the observer can occasionally verify (the LAD) generally features anteroseptal and anterolateral that both arteries are indeed subepicardial (instead of papil- (diagonal) branches as it courses along the anterior interven- lary muscles, penetrating coronary branches, or subendocar- tricular groove and tapers toward the cardiac apex. In some dial collateral vessels). Coronary crossing affects secondary instances, a large subepicardial anterior artery is not encoun- vessels and only rarely causes clinical problems, such as tered in the interventricular groove because one of the fol- difficulty in identifying a branch to be grafted during coro- lowing alternative patlerns is present: nary artery bypass. 1. The proximal anterior descending afieryloo':+s or a su- (See Case Reports) Report 4.37 in the Atlas of Case pernumerary aortic ostium2e6 gives rise to a single large first septal branch that supplies most of the secondary Anomalous Origination of the Posterior Descending anteroseptal branches, leaving a small or absent mid- Artery from the Anterior Descending Branch or a distal subepicardial LAD (which is improperly called Septal Penetrating Branch atretic). 2. The proximal LAD splits into two smaller, parallel According to a consistent rule coronary morphology, of branches ("split LAD"l,ttz which run along the ante- anterior septal penetrating branches do not reemerge on the rior interventricular groove. side of ventricular septum. In cases of posterior opposite the 3. A large diagonal artery originates quite proximally from descending artery occlusion, however, they are frequently a the LAD and runs parallel to it, giving rise to all the source collateral connection with the facing. posterior of anterolateral branches. Beyond the origin of the first septal vessels. Only rarely have cases been reported of an septal branch, the residual distal LAD is left with limited unusually large anterior septal branch that not only pene- dependent territory and appears as a very small vessel, trates the whole extent of the septum but also reappears, where a bypass graft could not typically be implanted. in a subepicardial position, in the posterior interventricular 4. A highly dominant RCA gives rise to most of the ante- groove? and produces the terminal portion of the posterior rior septal penetrating branches by producing an anoma- descending branch.ss'367 One might doubt the congenital na- lous LAD177 (via the direct intraseptals6 or the anterior ture of such an anomaly (versus an acquired occlusion of prepulmonicao'4l8 route, unusually prominent posterior the posterior descending artery with collateral circulation septal branches, or a wrap-around-the-apex posterior de- from an anterior septal branch), especially in the context of scending branch). coronary atherosclerosis. In a more common and clinically relevant pattern, the posterior descending branch originates In similar cases, the small size of the LAD might lead to congenitally from the distal LAD after encircling the cardiac the erroneous conclusion that the LAD territory is ischemic apex (see the next section). because of the unusual coronary pattern. 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ANOMALIES OF TERMINATION: CORONARY tions. Appearance of the plastic gel in a cardiac cavity was FISTULAS considered evidence of an arterio- or veno-cameral communication, An important but unproved condition for accurately As the coronary artery tree branches and propagates pe- establishing this diagnosis was that the plastic gel not get ripherally, it undergoes continuous tapering until it reaches past the capillary level. Using these techniques, Baroldi and the arteriolar level (the last segment of the coronary circula- Scomazzoni5os demonstrated 2O0- pt arterioluminal commu- tion to have a tunica media). The arterioles eventually drain nications in 867o of the left ventricles and 5O%o of the right into the capillary network, and only occasionally do they ventricles but not in the atria; they also demonstrated <2- maintain small communications with sinusoidal intratrabec- mm veno-cameral communications in the left ventricle, right spaces. A sizable communication between a coronary ular ventricle, and right atrium but almost never in the left atrium. (1) (2) any segment of the artery and a cardiac cavity or The relationship between these findings and angiographic pulmonary is generally called a coro- systemic or circulation observations or functional status is not immediately clear. a pressure lower than that nary fistula.3" Any structure with During angiography, only arterial injections are made, and aorta (i.e., equal to the proximal coronary of the systemic any communication with a cardiac cavity is visualized at the pressure) can drain blood flow from a coronary artery if an end of contrast runoff. Clinically, however, it is impossible abnormal communication occurs, allowing fistulous flow. In to know whether the capillary bed is or is not perfused by reviewing the anatomic literature, starting with the historic "fistulous" blood, which would differentiate potentially (1708), early reports ofVieussens (1706) and Thebesius Bar- damaging (arterio-cameral) communications from benign oldi and Scomazzoni5o8 analyzed recurrent, lively discus- (veno-cameral) ones. sions525 that have occurred over the centuries about coro- Only during RCA injections (especially when performed naro-cameral communications in the normal heart. These subselectively and under high pressure) is contrast material authors concluded that two types of communications nor- frequently observed to drain into the anterior right ventricle mally exist in the human heart, as alternatives to normal and the right atrium.r2s This finding is not to be confused drainage into the corona.ry veins/coronary sinus system: with the presence of thebesian veins. It merely shows that 1. Communications originating at the arteriolar level are coronary veins of the right atrium and ventricle often drain established indirectly by means of arterio-sinusoidal directly into these cavities, without communicating with the vessels (which are irregularly shaped, measure 50 to 250 coronary sinus. g,m in diameter, lack a tunica media, and drain into any Most likely, what appear as coronaro-cameral communi- >200-pc cardiac cavity), or directly by means of arterioluminal cations on angiography correspond to the anatomic normal hearts. vessels (which are 40 to 200 p,m in diameter, have a channels seen by Baroldi and Scomazzoni in to specific thin media, and drain into any cardiac cavity). Nevertheless, these channels should be subjected proper ofcoronaro- 2. Venous communications or thebesian veins (first de- studies, using techniques. The definition cameral fistulas is clear when larger vessels are involved, scribed by Thebesius 11686-11321, who injected var- but it is not totally clear when smaller, multiple vessels are ious substances into the coronary sinus) are direct com- involved.s6'170'343 angiographic fistulas tend to occur munications between a coronary vein and a cardiac 5*u11 only occasionally in patchy aggregates, affecting the smaller cavity. They are especially common in the right atrium ramifications of several adjoining coronary branches or ar- (where they measure tp to 2 mm in size) and in the teries, and typically involving the apical portions ofboth the right ventricle. left and right ventricles but never the outflow tracts. In such The exact nature of these small coronaro-cameral connec- instances, the flow pattern indicates the presence of multiple tions remains unclear. However, they should probably not small communications with limited flow, which is generally be called fistulas, because they do not involve substantial only diastolic into the left ventricle but systolo-diastolic into fistulous flow. the right ventricle.56'i7o These communications do not cause Angiographically, it is usually impossible to visualize the ectasia of the proximal feeding arteries, nor do they tend to smaller coronary artery to cardiac cavity communications enlarge with time.2el without the use of wedge injections.r28 Baroldi and Scomaz- This subject is being discussed extensively herein because zoni found such communications in almost all specimens by of widespread persistent ignorance about the exact nature of injecting the coronary arteries with latex or neoprene, which the smaller communications between corona.ry vessels and is fluid at room temperature but solidifies at 40 to 50 "C. cardiac cavities.332 The overwhelming current tendency in The injections were made at 200 mm Hg of continuous pres- the literature and in practice is to group these entities under sure and were followed by further manual compression to the general heading of coronary fistulas and to imply that enhance fluid progression during the 5- to l0-minute prepa- nutrient blood is shunted away from the myocardium.lll Nu- ration period. To visualize veno-cameral communications, merous reports have been published regarding patients with these investigators injected the coronary sinus with the same angina, ischemia, myocardial infarction, or arrhythmias in plastic material (of a different color), at 70 mm Hg of pres- the presence of multiple small, patchy coronaro-cameral sure, a few minutes after the beginning of the arterial injec- communications. 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nutrient myocardial flow, rather than simply to eliminate the is during the patient's fifth to fifteenth year of life; the timing fistulous tract. The absence of nutrient coronary branches of surgery in such cases should not be based on the time of arising from a fistulous tract should suggest an alternative onset of symptoms, the absolute amount of fistulous flow,38e diagnosis, namely ruptured aneurysm of an aortic sinus.447 or signs of congestive failure or myocardial ischemia during With the larger fistulas , alarge quantity of contrast medium stress testing. If the diagnosis is missed in the 5- to l5-year should be injected (about 20 ml/sec for at least 2 seconds), age range (as it frequently is), the indications for, and timing and the fistula should be examined in different projections, of, intervention may change because of variance of the risk- depending on its specific anatomy. It is prudent to avoid benefit ratio.ao6 Older patients445'4e5 may still do very well small-lumened catheters with only end holes (coronary pre- at surgery but may have more frequent complications (espe- formed catheters) with high injection pressure and to use cially peri- and postoperative myocardial ischemic events3o3' large-lumened catheters with side holes (like an NIH@ or a 311's33 and extracardiac complications); moreover, after nor- Gensini''.6 angiographic catheter). Alternatively, one may use malization of blood flow, their remaining risk of coronary a coronary angioplasty guiding catheter (large-lumened), thromboembolic disease in the excessively ectatic coronary kept in position by a 0.014-inch guidewire. segments will at least equal that of patients treated medi- The proximal tract of a fistulous coronary artery should cally.s33 The recent introduction of catheter devices for the be regarded as an atypical example of a "mixed trunk" obliteration of coronary fistulassT' I 6l'2 I 7'303'3 l'1'3 I s'368'1o2'441 rather than as a simple coronary artery (which is defined as will be further discussed in Chapter 5. a vessel that provides exclusively nutrient flow). Concomitant obstructive coronary disease, in either the Over the years, torrential flow will induce clinically im- affected vessel or an unaffected one, may be the most com- portant morphologic changes in the walls of a fistulous coro- mon reason for clinical recognition of a coronary fistula and nary artery. The fistulous tract-but not the distal, exclu- for surgical intervention in older patients. sively nutrient branches-will undergo progressive changes Fistulous coronary connections usually involve structures that range from simple dilation (as would be expected be- that adjoin the coronary arteries, such as the coronary veins cause of the increased blood flow) to frank aneurysm forma- and the four cardiac cavities. I I 8'237'338'381'406'43sAa3Aaa y""" 1iontg' r26.262.28a 3a2.s22 (reco gnizable only because dilation is frequently, in otherwise normal hearts, a coronary fistula greater than in the adjacent vessel), intimal ulceration, me- will drain into an extracardiac structure such as the pulmo- dial degeneration, intimal rupture, atherosclerotic deposi- nary artery or its main branches or the superior vena cava.18' tion, calcification,28s side branch (nutrient) obstruction,a3a 466 Coronary-to-main pulmonary artery fistulas are usually and mural thrombosis.a5'a3a'aa2 16" ultimate, dreaded but small, multiple,16 and of no clinical significance. Congenital rare, complication of the increased wall stress is coronary coronary-pulmonary connections are sometimes seen in the rupture into adjacent cardiac structures6l or the pericar- context of other congenital heart defects, especially critical dium.ls6 Because the vessel wall's reaction to the prolonged pulmonary valve stenosis or atresia or (even more fre- increased flow is so variable, the observer should be cautious quently) pulmonary branch stenosis or atresia, or coarctation in estimating the amount of fistulous flow on the basis of the aorta. of luminal diameter alone. Vessel size, in itself, may be a Although the literature contains frequent references to t7 t fallacious parameter: in the most extreme case, a very ' 'coronary-to-bronchial artery fistulas ,t; '41'141't7 '347 '366'373 large, aneurysmatic fistula could eventually become throm- we doubt the existence of such an entity. Both of these arter- bosed, totally obliterating fistulous runoff to the distal ies are, in fact, systemic in patients with a normal cardiovas- u"rr"1. 1 18,1s0,249,364 cular anatomy, and fistulous flow cannot be expected to In indicating whether intervention is necessary,3ee the occur between two vascular sites that have identical pressure amount of dilation of a fistulous vessel has recently become regimens. Communications have indeed been observed with more relevants2'118'333 than the amount of fistulous flow or special frequency between a coronary artery and a lung seg- symptoms and/or signs of myocardial ischemia. 1 1a'38e 6u15"- ment with a chronic infectionl (bronchiectasia, sequestra- ter-based26e or surgical63 intervention at an early age is gen- tion, or pseudosequestration); in these cases, the involved erally recommended for patients with large fistulas, because vessels behaved more like neovascularizations or collater- of the risk of rupture and mural clottinga3a'aaz; moreover, u1r22o3ae than like fistulas.lT'173 Only occasional reporters aortic sinus disruption caused by an extremely enlarged cor- have suggested that a "coronary to bronchial anastomosis" onary ostium can result in aortic insufficiency. Late athero- exists and can cause a myocardial infarcti on.17'27e In congen- sclerotic and thrombotic changes2lS will evolve even after itally sequestered pulmonary lobes, the systemic arterial sup- total obliteration of the fistula. With respect to the optimal ply is usually derived from the descending or abdominal timing of surgery, another major consideration should be aorta; in pseudosequestered lobes, however, it may originate that reversibility of the ectasia is consistently reported only from intercostal, mediastinal, subclavian, and pericardial ar- after surgical correction of fistulas in pediatric casesl I 8 and is teries, l7'173'24:'zue .6i"6 probably connect with pulmonary never observed in older patients.52 Because of these factors, arterial branches,2o not bronchial ones. many authorities have concluded that, once a large coronary Coronary-to-pulmonary communications may also appear fistula has been diagnosed, the optimal time for correction after cardiac surgery, even heart transplantation (see Case ur eseeJcur obog e :poseeJJur sr pueuep uo8^{xo eql 'peol go lueue8relue e,rrsserSo-rd yo usrueqceru eql eq ,(eu uorl ->lro,ry\ ernsserd pesuercur ue ro esrcrexe qll71\ '(o5y9) 4tolrl. -EIrposB^ pelerperu-^\old'(tuorulrrucer leJelulloc s€ u,rou>l ernsserd uuql f8reue ssel spueuep (o7og1) 4tortr eurnlon '.{1r sr ssecord slqt) tuerper8 ernsserd € Jo lesuo eql Jo llnser -Artce I€culoele ol peloêp sr. obI pue'(Ueeq pepeolun eq1 ur) e su e8myue puu uedo f1mo1s ,{eu slereleloc eseql 're,r.e tusqoqeletu pseq aql ol peleclpep sr lueruerrnber ue3,(xo prp -lroq 'uorsnlcco ,(reuoroc e let1e isuerur8er ernsserd eurus -reco,(ru eqt1o ob1z '.,(lssorg'(3 OOyutunu 911'6) elcsnur eql qll.&\ seueu? ueealeq ./r\ou Jo eruesqe IIe Jo llnser B sB Ielele>ls Jo 1uqt ueql (3 g6l/uru/-1ru 0t 01 8) releer8 qcnu sr pesolc eq 01 puol ,(lquqord sleretulloc (e1qrsr.r.ur ,(lpcrqderS 'pueru 'uorsnlcco unrpruco.{ur eql Jo lueruerrnber ue8.(xo eql 'lser lv -or8uu) Iurruou 'slenpr^rpur ,(q1peq uI ,fteuoroc -ep ueSfxo prpreco^{ur eLO lo ob06 o1 91 seqddns flururou luersuer1 snor,terd e lno eIru o1 elqrssodurt .{11uenbe,r; 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myocardial contractility, pressure work, or the heart rate or corona"ry vasodilator (adenosine or dipyridamole) stress/ causes almost a 5O7o increase in the oxygen demand. During testing results in a "high-flow/low-resistance" state, in exercise, the maximal workload is 3 to 4.5 times greater than which a proximal coronary stenosis becomes more evident. sl2 at rest; normally, this increased workload results in coronary Recent investigationssoe have suggested that the coro- flow that is 3 to 4.5 times greater than baseline flow. nary tree has a dual mechanism of vasodilation: endothe- The oxygen content of systemic arteries is about 80 vol%o, lium-dependent vasodilation of coronary vessels measuring whereas that of systemic and coronary veins is 60 vol%o and >200 pm in diameter and nonendothelium-dependent vaso- 5 vol%io, respectively. This finding reflects the unusually high dilation of arteriolae measuring < 150-200 trlm in diameter. amount of oxygen extracted by the myocardium under basal Nitroglycerin acts by means of the first type of mecha- (extraction conditions being submaximal at rest). nism,122 which essentially affects only the proximal, epicar- myocardial The capillary density is about 3500/mm2 dial coronary arterial tone (capacitance vessels), where it is (compared with 400/mm2 in the skeletal muscles); 50tol\Ea converted into its active form (nitric oxide) by the endothe- of the left ventricular myocardial capillaries are patent at lium.lee In contrast, adenosine directly stimulates smooth- rest, and essentially I00Vo are recruited during maximal muscle-cell A2 receptors in arlerioles (resistance vessels), workload conditions. without affecting capacitance vessels. Papaverine seems to The capillaries have a diameter of about 3 p during systole affect both capacitance and resistance vessels. Myocardial and 4 p, during diastole, evidencing the normal phasic in- perfusion imaging for determining coronary functional re- crease in intramural pressure. The intercapillary distance is serve is essentially based on these physiologic parameters. about 17 at rest, versus I I to 14 during maximal capil- /u ;r A coronary anomaly may produce physiologic dysfunc- lary recruitment. A normal adult myocardial cell has a trans- tion or clinical consequences under the circumstances listed verse diameter of about I 8 ;r (versus 50 p for adult skeletal in Table 4.6, as discussed in the literature1o'72'238.327 and muscle cells), but hypertrophic myocytes can increase that summarized in the following sections. diameter to 3O p,. Therefore, the surface for metabolic ex- changes is about 15 times larger in normal myocardial cells than in skeletal fibers. TYPES OF PATHOPHYSIOLOGIC MECHANISMS Compared with the subepicardium, the subendocardium AND/OR CLINICAL IMPLICATIONS normally has a similar basal flow rate but an increased wall This section discusses mechanisms reported to be in- stress, intramural systolic pressure, and oxygen demand; it volved in causing clinical manifestations or changes the also has a decreased maximal flow rate (coronary reserve), in clinical outlook for patients with congenital coronary anom- tissue oxygen concentration atrest, and venous oxygen satu- ration. alies. The clinical relevance of coronary anomalies has recently been recognized by the The major regulators of coronary blood flow are (1) the American Heart Association, the American intramural pressure, (2) the aortic (coronary) pressure, which College of Cardiology, and the American has an elective dependence on the diastolic mean pressure Academy of Pediatrics.s34 The fact that 24Vo of sudden deaths and time (as about 857o of the coronary blood flow occurs in athletes can be related to coronary anomaliess3a during diastole under basal conditions), (3) the myocardial underscores the importance of this subject. metabolic rate, (4) the parasympathetic and sympathetic nerves, (5) endothelial function (autocrine), and (6) blood Misdiagnosis of the Coronary Anatomy viscosity, which is greatly increased in polycythemia. Coronary stenosis does not affect the resting blood flow Unusual coronary anatomic patterns may be confusing t as until more than 90Vo of the luminal diameter becomes com- and easily misdiagnosed.es, ltrlit6iugnosis may adversely promised. The coronary reserve (maximal vasodilatory ca- affect the treatment strategy and outcome, as well as the pacity) is normal in the presence of 0 to 6OVo l,luminal naffow- patient's psychosocial status and insurability. The following ing, but it progressively decreases to }Vo when the degree conditions may be particularly hard to recognize and/or in- of stenosis approaches 90Vo.In the coronary circulation, vas- terpret. cular resistance is the sum of the proximal (subepicardial) and distal (arteriolar) resistance. Proximal resistance is negli- (Pseudo) Absence of a Coronary Artery gible for proximal stenoses of up to 80 to 907o, but it becomes severe for stenoses of more than 90Vo. Endogenous Labeling a coronary artery that is not visualized directly vasodilators (especially nitrous oxide) and pharmacologic or by means of collateral, retrograde circulation as "absent," vasodilators will electively affect distal arteriolar resistance "occluded," or "missing" is usually erroneous in the ab- in the absence of significant (>907o) proximal stenosis. sence of an acute myocardial infarction. Further studies, in- With stenoses greater than9OVo, the peripheral vasodilating cluding ascending (and descending) aorlography and selec- reserve is lost. tive subclavian or carotid angiography, with follow-through Under baseline conditions, the coronary system is subject to the mediastinum (possibly using digital subtraction tech- to a "low-flow/high-resistance" state. In contrast, exercise nology), should be performed in these cases. 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sNvT{nH NI sslf,f,Jr{V snorv7{oNv (INV TWI}ION E9 / ^uvNodo3 66 / Cnarrnn4 ischemia can also occur in anomalies like fistulas, which episodes of superimposed spasm,26s'266338 intravascular cause nutrient myocardial flow to compete with fistulous clotting,3'207'260'434'442'47o or accentuation of systolic naffow- flow (in a lower-resistance circuit, organized in parallel) ing in myocardial bridges, as brought about paradoxically under conditions of limited supply, as in the case of a single, by vasodilators.T 4'246 restrictive proximal coronary trunk. This phenomenon rarely In recent years, the literature has included many case re- l,so2 occurs in the usual coronary fistula; it is more often associ- poftS2s,26,2o8,272,2e8,384,3es,4t that testify to the ischemic ated with anomalous origination of a coronary artery from nature (in the dependent myocardial region) of adverse clini- the pulmonary artery, in which the collateral circulation orig- cal events (myocardial infarction,32'7e't13'486 chest pain,aa inating from the normal coronary arteries preferentially syncope,a6l ischemic cardiomyopathy,a6l or sudden death5s' tends to drain blood into the ectopic corona.ry ostium in the 7e'24o'241'1s3'40t'+6e; in patients with various coronary anoma- pulmonary artery, while bypassing the myocardiul 6"6.s0'206 lies. Angiographic and autopsy tLr4i".2s'32'208'3e3 (carried The dramatic improvement in left ventricular function that out some variable time after the ischemic insult was substan- is frequently seen after effective repair of this anomaly is tially resolved, even if by death) have consistently failed to clear evidence that a chronic hibernation state may exist reveal the critical coronary obstructions or occlusions that well beyond infancy.s:'z:'t74'te3'zt6'232'3t7'363 Despite earlier would be expected in such cases. The implication is that a statements to the contrary in the literature, primary myocar- transient obstruction occurred, but this phenomenon could dial ischemia cannot be assumed to occur routinely because not be well documented. If such an obstruction can resolve of an anomalous course alone,83'3oo specifically when a coro- that quickly, it must be caused by a coronary spasm in most nary afiery courses between the pulmonary artery and the cases. Platelet clot formation or full thrombosis is unlikely aoria.335'476 The simplistic notion that such an anomalous to be missed in angiographic or anatomic studies performed course could be subject to an external scissors-like compres- shortly after the event occurs.3'384 Provocative testing de- sion mechanism44's8'ls1'200'383'410 ir not generally sustainable signed to document coronary spastic potential must be more in the light of current information. Indeed, these patients widely performed in clinical practice, to elucidate the patho- usually do not have reproducible angina or ischemia during physiologic mechanism and prognosis, thereby facilitating stress testing, and they tend to live out their entire lives rational therapeutic decision-makiflg.'t32'1s3'211 Spasm is without any ischemic manifestation. Nevertheless, a few of probably involved in (l) most cases ofectopic coronary orig- 1 08' 1 88' them have ischemic events-usually sudden death,6s' ination from the aorta, with tangential orientation of the 236'2so'261'383'38s a myocardial infarction,26l or syncope-in proximal trunk and a slitlike orifice or ostial ridgs2s'zse'+to the absence of coronary thrombosis or obvious stenosis.s8' and (2) some muscular bridges (intramyocardial coronary 11o'2o8 5u.h patients may have a tangential slitlike ostium272' arteries), especially the more extensive ones, in which long, 322 or ostial i.dgesa7z or membranes,66'31e'40e but they rarely thick myocardial bands cover a proximal main coronary have a critical fixed stenosis.2l3 At the present time, routine trunk. It is likely, but not fully certain, that phasic bending surgical correction (generally by means of bypass grafting) of the intramyocardial coronary segment is a stimulus to cannot be recommended on the basis of such anomalies spasm-even more than is systolic narrowing i1."1L211'3se alone; it would, however, be appropriate for survivors of Provocative testing of endothelial dysfunction or excessive sudden cardiac death or transient ischemia who have a posi- spasticity occasionally yield positive results. I 32' I s3'21 I tive provocation test. Despite exceptions widely reported in Conceivably, a coronary artery that originates from the the literature, myocardial ischemia cannot usually be docu- opposite coronary sinus and courses between the aorla and mented by stress testing, whether the patient has an isolated the pulmonary arlery could behave in a peculiar manner (pre- coronary intramyocardial course,388 multiple corona.ro-cam- viously unreported), especially during or after exercise or t'2s8'29 1 eral micro-communication s,7 or coronary aneurysms prolonged athletic training, leading to relevant clinical reper- or ectasia. If these conditions indeed lead to myocardial is- cussions. We allude to stretching of the abnormal vessel chemia,26 it is generally because of additional acquired fea- against the aortic wall while that vessel is being subjected tures, as described in the following section. to the opposing forces of right and left ventricular dilation brought about by exercise. Indeed, during strenuous exercise, the cardiac output of young, trained athletes may in- Secondary, Episodic Myocardial Ischemia crease to 25 to 30 L/min: under these circumstances, the Paradoxically, many coronary anomalies that, in fact, arise dramatic increase in diastolic retum flow to the ventricles during the fetal period never produce clinical manifestations is only partially compensated by an increased heart ratel' a until many years later, if at all. One would expect that a major increase in diastolic volume also results, which must congenital disease, especially a potentially lethal one, would be accompanied by important increases in aortic pressure not allow for normal life (as it frequently does, in the pres- and wall tension (both of which greatly exceed the respective ence of negative stress tests), although it could suddenly pulmonary values) and also in right and left ventricular wall cause a catastrophic event. In most cases, the congenital tension. 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involving extreme overload of the right ventricle (pulmonary chemic damage occurs prenatally in coronary atresia but atresia with intact ventricular septu6l2e'1+z) or the left ven- neonatally in anomalous origination of a coronary artery tricle (aortic valve atresia;.63 The obstructive lesions have from the pulmonary artery. been attributed to shrinkage and, even more, to fibrous inti- Some amazing observations have been widely confirmed mal proliferalion.88,l2e These lesions occur in the presence in regard to the nature and behavior of chronic ischemia in of high-velocity collateral flow, which vents the overloaded pediatric patients with ALCAPA. Anatomically and histo- ventricular cavities. Surgical palliation of hemodynamic logically, this cardiomyopathy may involve cardiomegaly, right ventricular overload may delay the progression of coro- left ventricular dilation, myocardial hypertrophy, secondary nary pathology but also may eliminate critically important mitral insufficiency, changes associated with an acute myo- coronary collateral sources in the presence of severe proxi- cardial infarction, and various degrees ofinterstitial, patchy, (see mal obstructions Chapter 6). Single coronary artery is or diffuse fibrosis. After surgical correction of these anoma- generically not associated with an increased incidence of lies, myocardial function may be recovered, often to an atherosclerosis; should atherosclerosis occur in the common amazing extent. Not only are the results of nuclear myocar- however, trunk, the clinical consequences would be unu- dial perfusion studies dramatically improved or completely sually severe, because the dependent myocardial teritory normalized but cardiomegaly, mitral regurgitation, and left includes the whole heart and no collateral circulation can ventricular systolic dysfunction may totally disappear.asa In develop.262 these cases, recovery is similar to that observed after revascularization of critical left main lesions but is consistently Secondary Aortic Valve Disease more extensive. This phenomenon clearly reflects the presence of a hibernating myocardium. Additionally, it is inter- In patients with coronary anomalies, clinically important esting to note that myocardial hypertrophy is a probable aortic valve disease may result from ( I ) an independent con- result of ischemia in young persons, and myocardial reab- genital defect associated with coronary anomalies or (2) an sorption (possibly by means of apoptosis) may occur after acquired defect secondary to longstanding coronary anoma- the chronic ischemia has been resolved. lies, especially those involving greatly increased fistulous Diffuse cardiomyopathy is also observed in some cases flow and enlarged coronary ostia-primarily coronary fis- of anomalous origination of the LCA from the right anterior tula and ectopic origination of the LCA from the pulmonary cusp. In such cases, ischemia is probably the original cause artery. As previously noted, when a coronary ostium attains of the cardiomyopathy, but, as previously discussed, this a greatly enlarged diameter, the structure of the aortic cusp event must be caused by clinical or subclinical episodic ob- and the adjoining leaflet may be critically altered, resulting struction (spasm and,/or clotting), followed spontaneous in aortic regurgitation. When a corona.ry artery originates by revascularization.a6l these cases, revascularization ectopically from the pulmonary artery, it is the normal con- In can prevent further worsening but may produce tralateral coronary artery (the source of collateral and fistu- not a total recovery lous flow) that may cause such impairment of the aortic of myocardial function. valve. Volume Overload

Increased Risk of Bacterial Endocarditis A coronary fistula may cause cardiac enlargement and (diastolic) The earlier literature contains a few scattered reports con- volume overload by creating significant shunting cerning an increased risk ofbacterial endocarditis or endoar- of blood through the fistula. Depending on the degree of teritis in patients with coronary anomalies, especially coro- fistulous flow and the size of the recipient cavity or vessel, nary fistulas. This risk is probably related more to coexisting different cardiac cavities will be affected to varying degrees. aortic valve anomalies (even if their repercussions are ini- Coronary fistulas that drain into a systemic vein, coronary tially minor) than to the coronary anomaly itself. The current sinus, right-sided cardiac cavity, or pulmonary artery (in- extreme rarity of endocarditisaso under such conditions may cluding anomalous origination of a coronary artery from the be caused by extensive use of antibiotics in the general popu- pulmonary artery) will cause left-to-right shunting at sys- lation and routine prophylaxis for endocarditis in the pres- temic or near systemic pressures, resulting in volume over- ence of "heart murmurs," even in the absence of a definite load (with cavitary dilation and an increased diastolic work- diagnosis. load). Pulmonary hypertension is only rarely observed in cases involving coronary fistulas draining to the right ventricle or pulmonary artery, because the degree of shunting is Cardiomyopathy never excessives2 1i.e., greater than the systemic cardiac out- A cardiomyopathy is more likely to accompany ALCAPA put or entailing a pulmonary-to-systemic flow ratio of or left coronary atresia than anomalous origination of the >2: l). 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