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Case rePorTs in Cardiology
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Figure 2 Drawing of the heart in our 48-year-old man (DCMEO No. 82-03-291).
Abbreviations as in Figure 1.
major coronary artery within myocardium protects the intramyocardial segment
from atherosclerotic plaques.
REFERENCES
1. Gallavardin L, Ravault P: Anomalie d’origine de la coronaire anterieure. Lyon
Med 136:270, 1925.
2. Kintner AR: Anomalous origin and course of the left coronary artery. Arch Pathol
12:586, 1931.
3. Born E: Uber Missbildungen der Kranzarterien und ihre Beziehungen zu
Zirkulationsstorungen und plotzlichem Tod. Virchows Arch Pathol Anat 290:688,
1933.
4. Cheitlin MD, De Castro CM, McAllister HA: Sudden death as a complication
of anomalous left coronary origin from the anterior sinus of Valsalva. Anot-sominor congenital anomaly. Circulation 50:780, 1974.
5. Roberts JT, Loube SD: Congenital single coronary artery in man. Report of nine
new cases, one having thrombosis with right ventricular and atrial (auricular)
infarction. Am Heart J 34:188, 1947.
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Case 490 origin oF The leFT main From The righT Coronary arTery
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6. Allen GL, Snider TH: Myocardial infarction with a single coronary artery. Report
of a case. Arch Intern Med 117:261, 1966.
7. Snow PJD: Acase of single coronary artery with stereographic demonstration of
the arterial distribution. Br Heart J 15:261, 1953.
8. Moodie DS, Gill C, Loop FD, Sheldon WC: Anomalous left main coronary artery
originating from the right sinus of Valsalva. Pathophysiology, angiographic denition, and surgical approaches. J Thorac Cardiovasc Surg 80:198, 1980.
9. Bochdalek J: Anomaler Verlauf der Kranzartenen des Herzens. Virchows Arch
Pathol Anat 41:260, 1967.
10.
Sanes S: Anomalous origin and course of the left coronary artery in a child.
So-called congenital absence of the left coronary artery. Am Heart J 14:219, 1937.
11.
White NK, Edwards JE: Anomalies of the coronary arteries. Report of four cases.
Arch Pathol 45:766, 1948.
12.
Rossi L, Dander B, Nidasio GP, Arbustini E, Paris B, Vassanelli C, Buonanno C,
Poppi A: Myocardial bridges and ischemic heart disease. Eur Heart J 1:239, 1980.
13.
Morales AR, Romanelli R, Boucek RJ: The mural left anterior descending coro-
nary artery, strenuous exercise and sudden death. Circulation 62:230, 1980.
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Case 508 Crisscrossed Atrioventricular
Valves and Prolonged Survival
William C. Roberts, MD, Thomas L. Spray, MD,
Richard J. Shemin, MD and Barry J. Maron, MD
Normally, of course, the atrioventricular (AV) cardiac valves have a right-to-left relationship with one another (Figure 1). The right-sided AV valve connects the rightsided atrium to the right-sided ventricle, and the left-sided AV valve connects the
left-sided atrium to the left-sided ventricle. In rare cases, however, the AV valves
are not parallel to one another but are crisscrossed, so that the right-sided atrium
connects to the left-sided ventricle and the left-sided atrium connects to the rightsided ventricle (Figure 1). When this occurs, the AV valves have an anteroposterior
relation. Since 1974, a number of patients with crisscrossed AV valves have been
reported on, and most died early in life from either inadequate or excessive pulmonary blood ow, the latter by way of an associated large ventricular septal defect.
This report was prompted by studying a man who lived 55years with crisscrossed
AV valves associated with double outlet right ventricle and pulmonic valve stenosis.
Although cyanosis and a precordial murmur were present at birth and throughout life,
the patient was asymptomatic except on extreme exertion until age 46years when syncope
occurred; cardiac catheterization was performed, and operation refused. At age 54years (10
months before death), 2,500 ml of blood was removed because of a hematocrit level of about
65%. Two months later, diplopia, a sixth nerve palsy, and intermittent Mobitz II heart block
with a left bundle branch block pattern appeared, and cardiac catheterization was repeated.
The pressures (in mm Hg) were as follows: pulmonary artery, 15/7; right ventricular outow,
60/10, and inow, 135/7; right, atrial mean, 5; pulmonary artery wedge mean, 7; left ventricle, 125/10; and aorta, 125/71. Examination 4 months before death disclosed a grade 3/6
precordial systolic murmur with thrill, most prominent at the left sternal border. The electrocardiogram, chest roentgenograms, and M-mode echocardiogram are shown in Figures2
to 4. The right subclavian artery was anastomosed to the right pulmonary artery 70 days
before death. Immediately after operation, intractable congestive heart failure developed and
6 days later the Blalock-Taussig shunt was closed; his condition improved briey, but gradually evidence of poor perfusion of the kidneys, heart, and brain developed and the patient died.
The ndings at necropsy are summarized in Figures 1, 5, and 6. The mitral valve,
located posterior to the tricuspid valve, connected the right-sided anatomic right atrium
to the left-sided anatomic left ventricle, and the tricuspid valve, located anterior to the
mitral valve, connected the left-sided anatomic left atrium to the right-sided anatomic right
ventricle. The only outlet for blood from the left ventricle was a ventricular septal defect.
Both aorta and pulmonary trunk arose from the right ventricle. The pulmonic value was
severely stenotic and heavily calcied (Figure 6), and a portion of its sinus wall protruded
anteriorly (Figure 1).
From the Pathology, Surgery, and Cardiology Branches, National Heart, Lung, and Blood
Institute, National Institutes of Health, Bethesda, Maryland. Manuscript received and
accepted July2, 1982.
Address for reprints; William C. Roberts, MD, Building 10A, Room 3E-30, National Institutes
of Health, Bethesda, Maryland 20205.
204 DOI: 10.1201/9781003409342-30
Case 508 CrissCrossed aTrioVenTriCular ValVes and Prolonged surViVal
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Figure 1 Diagram showing basic arrangement of the patient’s crisscrossed heart
(top) compared with the normal heart (bottom). The valves shown on the right are
as they appeared after removing the aorta (Ao), pulmonary trunk, and walls of the
right atrium (RA) and left atrium (LA). The ventricular septum in the crisscrossed
heart is relatively perpendicular to the atrial septum, and this feature allows the
crisscrossed valve arrangement. Blood traversing the tricuspid (T) valve from the
posteriorly located LA ows anteriorly to the anteriorly located right ventricle
(RV) and blood traversing the mitral (M) valve ows posteriorly to the posteriorly located left ventricle (LV). The pulmonic valve (P) (top only) is unicuspid and
unicommissural, and an aneurysm (An) of its sinus protrudes anteriorly. L=left;
N= noncoronary cusp or sinus; P= posterior sinuses of Valsalva of the aortic
valve; R=right.
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Figure 2 Electrocardiogram showing intermittent second-degree heart block, left
bundle branch block, a P-R interval of 0.21 second, and lack of development of a signicant r wave in the precordial leads. V leads ½ standard.
Figure 3 Posteroanterior radiograph (left) and lateral angiogram (right). The
aorta (Ao) arises anterior to the stenotic pulmonic valve. LV=left ventricle; PT=pulmonary trunk; RV=right ventricle.
Figure 4 M-mode echocardiogram interpreted retrospectively showing (left) the
mitral valve (MV) to occupy most of the left ventricular cavity (LV), and (right) the
calcied pulmonic valve (PV) located anterior to the dilated left atrium (LA) and
posterior to the aorta. RV=right ventricular cavity; VS=ventricular septum.
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Figure 5 Anteroposterior cut of the heart showing (left) the right atrium (RA)
draining into the left ventricle (LV) and (right) the left atrium (LA) draining directly
into the right ventricle (RV). Both the aorta (Ao) and the pulmonary trunk arise
from the right ventricle. The tricuspid valve is displaced toward the right ventricle,
and its leaets partly obstruct the ventricular septal defect. The conus is inverted.
PV=stenotic pulmonic valve.
Figure 6 Radiograph of heart specimen disclosing heavy calcic deposits in the
stenotic pulmonic valve (PV).
This case is by far the oldest reported thus far with crisscrossed AV valves. Of
the other 9 necropsy cases reported,
the rst 2 months of life, 1 at 4years,
5]) and 1 at 25years (
20years and our patient had obstruction to pulmonary blood ow, a near necessity
for prolonged survival with transposition of the great arteries and ventricular septal
defect or double-outlet right ventricle.
1–6
4 (1[Case 13],
3
5
[Case 1]). Both of the 2 reported patients living longer than
2 at 8years (1[Case 12],4), 1 at 23years (6[Case
2,5
[Case 2],6[Case 7]) died during
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Figure 7 Transposition (Tr) of aorta and pulmonary trunk. Aproposed classication of transposition complexes including both parallel and crisscrossed AV valves.
Abbreviations as before.
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Nearly all patients with crisscrossed AV valves have malformed or transposed
great arteries, the most common being complete transposition and double-outlet,
right ventricle. Because most present classications of the transposition complexes
assume that the relation of the AV valves to one another is parallel, it is important
to delineate this relationship as parallel or crisscrossed because this factor affects
intracardiac blood ow and operative therapy
7
(Figure7).
REFERENCES
1. Lev M, Rowlatt UF. The pathologic anatomy of mixed levocardia. Areview of
thirteen cases of atrial or ventricular inversion with or without corrected transposition. Am J Cardiol 1961;8:216–263.
2. Anderson RH, Shinebourne EA, Gerlis LM. Criss-cross atrioventricular rela-
tionships producing paradoxical atrioventricular concordance or discordance.
Their signicance to nomenclature of congenital heart disease. Circulation
1974;50:176–180.
3. Symons JC, Shinebourne EA, Joseph MC, Lincoln C, Ho Y, Anderson RH.
Criss-cross heart with congenitally corrected transposition: Report of a case with
d-transposed aorta and ventricular preexcitation. Eur J Cardiol 1977;5(6):493–505.
4. Anderson KR, Lie JT, Sieg K, Hagler DJ, Ritter DG. Davis GD. Acriss-cross
heart. Detailed anatomic description and discussion of morphogenesis. Mayo
Clin Proc 1977;52:569–575.
5. Coto EO, Wilkinson JL, Dickinson DF, Rulantchas J, Marquez J. Gross distor-
tion of atrioventricular and ventriculoarterial relations associated with left juxtaposition of atrial appendages. Bizarre form of atrioventricular criss-cross. Br
Heart J 1979;41:486–492.
6. Van Praagh S, LaCorte M, Fellows KE, Bossina K, Busch HJ, Keck EW, Weinberg
PM, Van Praagh R. Superoinferior ventricles: Anatomic and angiocardiographic
ndings in ten postmortem cases. In: Van Praagh R, Takao A, eds. Etiology
and Morphogenesis of Congenital Heart Disease. Mount Kisco. NY: Futura,
1980:317–378.
7. Danielson GK, Tabry IF, Ritter DG, Fulton RE. Surgical repair of criss-cross heart
with straddling atrioventricular valve. J Thorac Cardiovasc Surg 1979;77:847–851.
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Case 534 Massive Right Ventricular Outflow
Tract Aneurysm After Ventriculotomy for
Subvalvular Pulmonic Stenosis Associated With
Peripheral Pulmonary Arterial Stenoses
Jeffrey E. Saffitz, MD, PhD*, Charles L. McIntosh,
MD, PhD and William C. Roberts, MD
Aneurysms that develop at sites of cardiac ventriculotomy incisions are recognized
but infrequent consequences of such procedures.
1
Their development appears to be
dependent on 2 major factors: (1) the peak systolic pressure within the ventricle after
the incision, and (2) the length of the incision. The incidence of postventriculotomy
aneurysm is far greater in the left ventricle than in the right because of the differences in peak pressures in each chamber. Herein, we describe a patient with both
subvalvular pulmonic stenosis and peripheral pulmonary arterial stenoses in whom
a massive right ventricular (RV) outow tract aneurysm developed after operative
relief of the subvalvular obstruction.
A 30-year-old woman (CC #05-28-81) had a precordial murmur at birth and cyanosis
and clubbing of the digits by age 7 years. Results of 4 cardiac catheterization studies, summarized in Table1, conrmed the presence of subpulmonic obstruction, peripheral pulmonary
arterial stenoses, and a right-to-left shunt through a patent foramen ovale. Syncope occurred
at age 12years, and by age 15 had become frequent and severe. The subpulmonic stenosis
was operatively relieved at age 15years by excision of portions of crista supraventricularis
myocardium. ATeon® fabric patch was used to enlarge the RV outow tract. The patent
foramen ovale was sutured closed.
Six months after operation, a pulsatile mass was present at the upper left sternal border
of the heart (Figure 1). During the next 13 years, the patient had no physical limitations
and received no medications (functionally class 1). She died suddenly at age 30years after a
febrile illness with hemoptysis.
Table 1: Hemodynamic data
Age (yr)
Pressures (mm Hg)
Pulmonary artery (s/d) 100/ 75/5 115/20 120/60
Right ventricle (s/d) 175/ 180/7 120/20 120/16
RV inow-outow peak
systolic gradient
Systemic artery (s/d) ... 110/60 120/60 120/70
PO=postoperative; s/d=peak systole/end-diastole.
75 105 5 0
Operative
8 15 Intra-
(6 mo PO) 16
From the Pathology and Surgery Branches, National Heart, Lung, and Blood Institute,
National Institutes of Health, Bethesda, Maryland. Manuscript received and accepted
January4, 1983.
* On leave from the Department of Pathology, Washington University, St. Louis, Missouri.
210 DOI: 10.1201/9781003409342-31
Case 534 massiVe righT VenTriCular ouTFlow TraCT aneurysm
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Figure 1 Radiographs of the chest 6 months and 13years after operation, illustrating the development of the RV outow tract aneurysm. Athin rim of calcium lines
the wall of the aneurysm. PO=postoperative.
Figure 2 Diagrams illustrating various components of peripheral pulmonary
arterial stenoses and RV infundibular obstruction, and the consequences of the right
ventriculotomy. Ao=aorta; LV=left ventricle; PT=pulmonary trunk; RV=right
ventricle.
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