PATHOLOGIC ANATOMY OF COMPLETE TRANSPOSITION OF THE ARTERIAL TRUNKS

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PATHOLOGIC

ANATOMY

OF

COMPLETE

TRANSPOSITION

OF

THE

ARTERIAL

TRUNKS

Maurice Lev, M.D., Victor M. Alcalde, M.D., and Thomas G. Baffes, M.D.

Cng(’Ilit(Jl heart i)isease Research and Training Center, Hektoen Institute, and the Departments Of

P(:tIiologt/ 011(1 Surgcrsj of tile Children’s Memorial Hospital, and the Departments- of Pathology of Northwestern Unieersity Medical School and the

Unicersity of Chicago School of Medicine

This investigation was supported by Research Grant (H-3351) from the National Heart Institute, Public

Health Service.

ADDRESS: (ML.) Hektoen Institute, 629-637 South Wood Street, Chicago 1.2, Illinois.

PEDIAnucs, August 1961

293

I

PREVIOUS COMMUNICATIONS, the

patho-logic anatomy of transposition

com-plexes, including complete transposition,

were classified and discussed.15 Complete transposition has also been discussed by other authors.613 This report deals with an

analysis of 70 cases of complete

transposi-tion studied from the museum specimens of

the Children’s Memorial Hospital and 77

cases seen at the Congenital Heart Disease

Research and Training Center. This analysis

has resulted in what we believe is a

clanifi-cation of the subject for the pathologist. This study is limited to cases of complete

transposition in pure levocardia. It does not

deal with cases in mixed levocandia

(in-verted transposition-corrected

tnansposi-tion) or in dextrocardia. In pure levocardia, transposition may be defined as an abnon-mality in the position of the aorta on its

remnant with respect to the pulmonary

artery or its remnant, and with respect to the chambers from which they arise. Com-plete transposition may thus be defined as the emergence of the aorta from the right ventricle and the pulmonary artery from the left. Thus it does not include the Taussig-Bing syndrome. The term can include

pul-monary atresia, where the aorta comes out completely from the right and the pul-monary remnant has a position reminiscent

of complete transposition.

The cases of complete transposition were

studied according to position and size of

the arterial trunks, relative sizes and

thick-ness of walls of the atnia and ventricles,

nature of septal defects, architecture of

the muscle bundles of the right ventricle,

and associated abnormalities. The absolute

sizes and muscle mass of the chambers, as

compared to the normal, and the

distribu-tion of the coronary arteries, are not in-cluded in this work and will be reported separately.

CLASSIFICATION

The classification in Table I was adopted for the 147 cases studied morphologically.

The age incidence for the individual types

was studied for 123 cases that came to necropsy and is included in the table. It can be seen that transposition of the arterial trunks is largely a pediatric problem. It is surprising, however, that some patients sun-vive to their teens.

Complete Transposition with Normal Archi-tecture without Ventricular Septal Defect

(51 Cases)

In complete transposition with normal

architecture without ventricular septal de-fect, the architecture of the ventricles was deviated only slightly from the normal. Yet, the aorta emerged from the night ventricle and the pulmonary artery from the left. Usually, the aorta was situated anteriorly and to the right, and the pulmonary artery posteriorly and to the left (Fig. 1).

Occa-sionally the aorta was situated anteriorly

and the pulmonary artery posteriorly (Fig.

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pos-tenor and left anterior sinuses of Valsalva (Fig. 4). The tricuspid valve was either

nor-(

Fig. 4). In most cases there was a charac- mally formed, or there was a slight ab-teristic type of crista; the septal band was

normal; the panietal band was broad and

sharply demarcated from the septal band; the parietal band formed most of the cnista

FIG. 1. Complete transposition with normal

arch-tecture. The anterior surface of heart is shown

with the usual position of arterial trunks; the aorta

(A) is anterior and to the right and the pulmonary artery (P) posterior and to the left.

Fic. 2. Complete transposition with normal

archi-tecture. The anterior surface of heart is shown with

one of the unusual positions of the arterial trunks;

the aorta (A) is anterior and the pulmonary artery

(P) posterior.

3. With pulmonary stenosis 7 5 11 days-74 yr yr 9 mo

B. With common ventricle (cor biatriatum triloculare) 4 Sf yr-1 yr 1 yr mo

C. With single ventricle and small outlet chamber’4 7 6 8 days-b yr 3 yr 44 mo

II. YSITH AIINORMALA-V ORIFICE

A. With tricuspid stenosis or atresia 11 13 Stillborn-44 yr 1yr 8 mo

B. With mitral stellOsis or atresia 5 5 3 days-4 yr 9 mo wk

C. With common A-V orifice 8 5 3 days-8 yr I yr 8f mo

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h ,

v’

Fic. :3. Complete transposition with normal

archi-tecture. The anterior surface of heart is shown, \vitll another ullusual position of the arterial trunks;

the aorta (A) is to the right and the pulmonary artery (P) to the left.

In complete transposition with normal

architecture with ventricular septal defect, the architecture of the right ventricle was

somewhat more disturbed than in the previ-ously discussed complex. However, it still

FIG. 4. Complete transposition with normal archi-tecture, right ventricular view, showing coronary

ostia (C), panietal band (Pa), septal band (S), and

usual architecture of the cnista.

Fic. 5. Complete transposition with normal

archi-tecture, left ventricular view, showing defect (D)

of the ventricular septum situated below the

pul-monary artery (P), between the anterior and

p05-tetior septa.

normality in the connections of the medial leaflet. This was connected by small papillary muscles to the septum.

In almost all cases there was either a probe-patent or widely patent foramen

ovale, and in most cases there was a patent

ductus arteniosus. The pulmonary artery was larger than, equal to, or smaller than

the aorta. Usually, the right and left ventri-des were of equal size and thickness, on the right ventricle was larger and thicker than the left. Less frequently, the left ventricle was larger and thicker than the night. The same applied to the atria. There were fre-quent abnormalities of the eustachian and thebesian valves. In a few cases, there was a bicuspid or otherwise abnormal pulmonic valve, a fetal coarctation, or a left superior

vena cava draining into the coronary sinus.

Complete Transposition with Normal

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FIG. 6. Complete traIlSl)OsitiOfl vith normal

of the ventricular septum, situated some distance l)elOW the lt1lmo11tr> artery (P) between the

an-terior and posterior septa.

FL,. ,.. .,)1ete transposition with normal

archi-tecture, right ventricular view, showing defect (D)

of the ventricular septum excavating the septal band (S), and the panietal band (Pa).

bore a strong resemblance to the normal. The position of the arterial trunks was the

Fic. 7. Complete trans]position with normal

of the ventricular septum, situated in the posterior

septum at the base, and the pulmonary artery (P)

same as in the previous type, as were the

coronary ostia. The ventricular septal de-fect was either in the anterior septum at the base (Fig. 5), at the junction of tile anterior and posterior septa at the base (Fig. 6), in

the pars membranacea, or in the posterior

septum at the base (Fig. 7). Uncommonly

the defect was in a more apical portion of the septum. In a few cases there were two defects, one at the base and one near the

apex, or both were at the base, one in the anterior and the other in the posterior sep-tum. Accordingly, the defect entered the right ventricle, either without disturbing the cnista or altering the cnista in various ways.

Where the defect was in the anterior sep-tum, it usually opened into the right

ventri-dc beneath the cnista, or the cnista was

excavated mildly or to the point of complete

separation of the septal and parietal bands.

\Vhere the defect was between the anterior

and posterior septa, or in the posterior

septum, it usually altered the septal band

(Fig. 8). Commonly, tile defect was unre-lated to the orifices of the aorta and

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Fic. 9. Complete transposition with pulmonic ste-nosis, left ventricular view, showing the subpul-monic stenosis (ST) and the ventricular septal

defect (D), and pulmonary artery (P).

FIG. 10. Complete transposition with pulmonic stenosis, right ventricular view, showing defect (D)

adjacent to aorta (A).

Fic. 1 1. Complete transposition with pulmonary

atresia, left ventricular view, showing aorta (A),

closing ventricular septal defect (D), and

atresia (At).

to tile pulmonary artery than the aorta, but

in some cases it was related to both. The

relative sizes of the aorta and pulmonary

artery varied. The tricuspid valve was more

often abnormally formed than in the

previ-Otis type; the medial leaflet was connected

to papillary muscles or to the rim of the defect.

The foramen ovale was usually open, but the cluctus arteniosus was often closed. A few cases showed closure of both. In most

cases the pulmonary artery was larger than

tile aorta. Uncommonly, the vessels were of

equal size, or the aorta was larger than the pulmonary artery. In the majority of cases, the left ventricle was larger than the right

ventricle; but in many cases, the right

yen-tricle was larger than the left, and in a few

cases they were equal in size. In general,

tile ventricles were of equal thickness; but, in some cases, the night was thicker than

the left. However, it was uncommon for the left ventricle to be thicker than the night. In most cases, tile right atrium was larger than the left; in a minority, both atnia were of equal size or tile left was larger than the

right. In general, both atria were of equal tilickness, or the right was thicker than the

left. Uncommonly, the left atrium was

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FIG. 14. Complete transposition with single ventricle

and small outlet chamber, showing aorta (A),

ptl-monary artery (P) and small outlet chamber (0).

In cases of complete transposition with

I . #{149}-‘

FIG. 13. Complete transposition with common

yen-trick, showing tricuspid orifice (T), mitral on-fice (M), pulmonic orifice (P) and aortic orifice (A).

FIG. 12. Complete transposition with common ventricle and aortic hvpoplasia, anterior view,

showing aorta (A) and pulmonary artery (P).

tilebesian valves were common. Other as-sociated abnormalities were bicuspid

pul-monic valve, bicuspid aortic valve, fetal

co-arctation, displaced right atrial appendage,

cleft aortic leaflet of the mitral valve,

con-genital aneurysm of the pars membranacea with the defect, and double aortic arch.

Complete Transposition with Normal Archi-tecture with Pulmonary Stenosis (7 cases)

normal architecture with pulmonary

ste-nosis, the architecture of both the right and left ventricles deviated more from the

nor-mai than in the two types just discussed (Figs. 9 and 10). Yet, the basic architecture of the ventricles was retained. The

pul-monary artery and aorta were situated

an-teniorly-posteniorly, respectively, or the aorta was anterior and to the right, and the

pulmonary artery posterior and to the left.

The coronary ostia were situated either in the posterior sinuses of Valsalva, in the pos-tenor and right anterior, or in the posterior and left anterior. The ventricular septal de-fect was in the anterior septum, or at the junction of the anterior and posterior septa. The defect was not confluent with the mouth of the pulmonary artery (Fig. 9), but in some cases it was confluent with the mouth of the aorta (Fig. 10). The defect opened into the right ventricle, excavating the cnista (Fig. 10), in the cases presented here. One case in this series presented no defect. Another case showed pulmonary atresia,

with the remainder of the heart being identical in type with tile others (Fig. 11).

The left ventricle, in this series, presented a separate narrow outflow tract, which gave rise to the pulmonary artery (Fig. 9). The

lining of the tract showed marked

fibro-elastosis. Tile pulmonary orifice was

nan-rowed or normal. The pulmonary artery was

smaller than the aorta, or they were the

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FIG. 15. Complete transposition with single ventricle

and small outlet chamber, with view of the right FIG. 18. Complete transposition with mild tricuspid

atrium anti single ventricle. stenosis (tricuspid valve, at T).

of the former. The pulmonary valve in some cases was bicuspid, or abnormally formed,

with marked hemodynamic changes. In the cases presented here, the left ventricle was

larger than the night (Fig. 9). The walls

were either of equal thickness, or the right or left dominated in thickness. An atrial

FIG. 16. Complete transposition with single ventricle

and small outlet chamber, with view of the left atrium and single ventricle.

FIG. 17. Complete transposition with single ventricle

and small outlet chamber, showing ridge (R) that

bears I)tllldle of His and bundle branches, and Fic. 19. Complete transposition with severe

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FIG. 20. Complete transposition with tricuspid ste-nosis, right ventricular view, showing right

ventni-cle (RV) and aorta (A).

F.. . omplete transpo tnic

atresia, left ventricular view, showing pulmonary artery (P), defect (D) of ventricular septum,

due-tus anteniosis (Du) and aorta (A).

Complete Transposition with Common Ven-tricle (4 Cases)

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septal defect was found, but the ductus was usually closed. Associated abnormalities in

this series were displaced right atrial

ap-pendage and night aortic arch.

FIG. 21. Complete transposition with tricuspid atresia, night atrial view, showing limbus (L) and

atresia (A).

In the complex of complete transposition with common ventricle (con biatniatum

tniloculare) (Figs. 12 and 13), there was an absence of the ventricular septum (Fig. 13), the only remnant being a low ridge on the

posterior wall, representing the posterior

septum. The latter carries the atrioventricu-lar bundle and both bundle branches. Both the mitral and tricuspid orifices entered, and both the aorta and pulmonary artery emerged from, the common ventricle. The

entrance into the aorta was in some cases

surrounded by a circular rim of

muscula-tune. In this series the aorta was situated an-tenionly and to the left and the pulmonary

artery posteriorly and to the right. The

aorta and pulmonary artery were equal in

size, or either vessel was larger. Pulmonary stenosis was present in one case.

Complete Transposition with Single Ven-tricle and Small Outlet Chamber (7 Cases)

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301

FIG. 24. Complete transposition with tricuspid

atresia, showing displaced right atnial append-age (RA), left atnial appendage (LA) and arota (A).

Complete Transposition with Tricuspid Atresia or Stenosis (1 1 Cases)

FIG. 23. Complete transposition with tricuspid atresia. right ventricular view, showing right

ventri-dc (RI’) and aorta (A).

FIG. 25. Complete transposition with mitral and

pulmonic stenosis and common ventricle, anterior view, showing the aorta (A).

outlet chamber 14 (Figs. 14-16) resembled

the preious type. Internally, however, the muscle bun(lleS of tile flgllt ventricle were

represented by a circular ring of muscle,

pro-ducing a pseudoseptum that separated a

main cllalTflber from a small chamber usually

situated anteriorly and superiorly. There

vas a small defect in this pseudoseptum. A

remnant of posterior septum was always

present on the posterior wall carrying the

conduction system (Fig. 17). The aorta was

situated anteriorly and to the left, and the

pulmonary artery posteriorly and to the right (Fig. 14), or tile vessels were

anterior-posterior. The main chamber received the

mitral and tricuspid orifices (Figs. 15 and

16) and gave rise to the pulmonary artery.

The small chamber gave rise to the aorta

(Fig. 14). The pulmonary artery was larger

than tile aorta. The coronary ostia were in

the posterior sinuses of Valsalva, or the

pos-tenor and right anterior sinuses of Valsalva.

The mitral and tricuspid valves were often

altered and in several cases difficult to

iden-tify, as were the inflow tracts into the single

ventricle.

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FIG. 26. Complete transposition with mitral and pulmonic stenosis and common ventricle, internal view,

showing aorta (A), pulmonary artery (P) and tricuspid valve (T). The pointer is in the mitral orifice.

diminutive night ventricle with a lange left

ventricle, or a single ventricle with small

outlet chamber. Under these circumstances

FIG. 27. Complete transposition with mitral and

pulmonic atresia, left atnial view. The arrow points

to the region of mitral atresia.

the ventricular septal defect was relatively small, and the aorta was smaller than the

pulmonary artery. In a variant (not seen in

this series, but previously seen by one of us {M.L.}) tile right ventricle or conus was divided from the main chamber by an ab-normal cnista with a large defect, and the aorta was large with pulmonary atnesia.

In this series, the aorta and pulmonary artery were situated anteroposteniorly, or the aorta was anterior and to the left and the pulmonary artery posterior and to the right, or the aorta was anterior and to the right and the pulmonary artery posterior and to the left. The coronary ostia lay in the

posterior, on posterior and right sinuses of Valsalva. In most cases the ventricular

sep-tal defect was small, and situated either in the posterior septum, on at the junction of the anterior and posterior septa, or in the anterior septum. It was usually not

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ri

FIG. 28. Complete transposition with mitral and

pulmonic atresia, ventricular view (may be

in-terpreted as single ventricle with separate conus,

or common ventricle), showing aorta (A), muscle ridge (Al) dividing off “conus” from ventricle, left inflow tract of ventricle (L), right inflow tract of ventricle (R) and ridge (Ri) probably carrying

atnioventnicular bundle and bundle branches.

FIG. 29. Complete transposition with common

atrioventricular orifice and pulmonary stenosis, right

atrial and right ventricular view. The arrows point

to the proximal and distal margins of the combined

septal defect.

atnioventricular orifice and pulmonary stenosis, right ventricular view, showing aorta (A) and accessory

ventricular septal defect (D).

of the fossa ovalis, a displaced right atnial

appendage (Fig. 24) and right aortic arch.

Complete Transposition with Mitral

Steno-sis or Atresia (5 Cases)

The complex of complete transposition

with mitral stenosis (Figs. 2 and 26) or atresia (Figs. 27 and 28) presented either a common ventricle or a single ventricle and small outlet chamber. The aorta and pul-monary artery were situated

antenopos-teriorly, or the aorta was to the right and

anterior and the pulmonay artery to the left

and posterior, or the aorta was to the left and anterior and the pulmonary artery to

the right and posterior. Pulmonary stenosis or atresia was present in all our cases. The left atrium was smaller than the right, and

there was an atrial septal defect of the

secundum type in all cases. An associated

abnormality was right aortic arch.

Complete Transposition with Common Atrioventricular Orifice (8 Cases)

In complete transposition with common

atrioventnicular orifice (Figs. 29-33), the

position of tile aorta and pulmonary artery

was anterior-posterior, or the aorta was

an-tenor and to the night, and the pulmonary artery posterior and to the left. The

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l LA

,

L.

i

FIr.. 31. Complete transposition with common

atnio-ventricular orifice and pulmonary stenosis, left “entnicular view, showing pulmonary artery at P.

atrio-ventricular orifice and pulmonary atresia, showing

aorta (A) and combined septal defect (arrows).

component. There was either a night and

left ventricle, slightly subdivided, with the right larger than the left, on there was either a single ventricle with small outlet chamber

or a common ventricle. The night atrium

was larger than the left. In all cases in this

FIG. 33. Complete transposition with common

atrio-ventricular orifice and pulmonary atresia, showing

aorta (A), pulmonary artery (P) and left atnial

appendage (LA).

series there was pulmonary stenosis or

atresia.

COMMENT

It is self-evident that complete transpo-sition as discussed above includes various entities that differ markedly physiologically and clinically. However we believe that from a pathologic standpoint this concept of complete transportation is helpful at the

necropsy table and may also prove advan-tageous from the angiocardiographic, cine-angiocandiographic and noentgenologic standpoints. Certainly from the

stand-point of embryology this grouping has a

unity, which has been discussed

else-where.1’2’ 7,11,15

It may be noted, as originally pointed

out by Spitzen,7 that this group (Groups 3 and 4 of Spitzer) presents the simplest

to the most complicated complexes. Thus in complete transposition with normal

archi-tectu.re, aside from the shunts, there are

only minimal variations from the normal architecture of the heart. On the other hand complete transposition with common

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chamber, show marked departure from the normal arcilitecture. Thus severe types of

transposition may be associated with

nor-mal or almost completely absent ventricular

septation.

It may be further noted that complete

transposition is often associated with

pul-monary stenosis or atresia. This is seen in a

few cases Witil normal architecture, is more

common in common ventricle, and becomes

very common in complete transposition

with mitral stenosis or atresia, or with common atrioventricular orifice. Complete transposition with common ventricle, with single ventricle and small outlet chamber,

and with tricuspid atresia are more often as-sociated with increased pulmonary flow.

The complex complete transposition \vitil

pulmonary stenosis deserves further

com-ment. Tile stenosis is almost if not always

some distance from the pulmonary orifice

and may be called “subpulmonary” or “left infundibular.” This stenosis may

pen-ilaps be acquired hemodynamically, due to

tile distance of the ventricular septal defect

from tile pulmonary artery. This is further

indicated by a case of pulmonary atresia

and what appeared to be a closing defect.

It may be postulated that this was originally a stenosis that went on to atnesia due to the diversion of flow through the defect and the fibroelastic reaction to the turbulent

flow.

There is a group of cases that may be

called partial or complete transposition with

pulmonary atresia. In these cases, the aorta

comes from the right ventricle, and the position of the aorta and pulmonary rem-nant, the coronary ostia, and the architec-tune of the muscle bundles of the right

ventricle are reminiscence of complete or partial transposition. There is always a ventricular septal defect in the anterior

sep-tum, and right ventricular hypentrophy. These cases are best considered under the concept of pseudotruncus, which also in-cludes over-riding aorta with pulmonary

atnesia, and they are therefore not con-sidered in this paper. We believe the term pseudotruncus should apply only to those

cases of over-riding aorta, partial or

com-plete transposition with pulmonary atresia, without other basic abnormalities such as absence of the ventricular septum, and ab-normalities in the atnioventnicular orifices, which are described above. Likewise

pul-monary atresia without a transposition

coni-plex should not be included in this category. It is also clean that complete transposition with common ventricle and with single vei’-tide and small outlet chamber are akin. If the muscle bundles of the crista are so

arranged as to produce a separate small outlet chamber, then the latter is obtained.

If on the other hand they are so situated as to produce a demarcation between the aorta and pulmonary artery, without

actually setting off a small outlet chamber, then the former is obtained. Sometimes the musculature is so arranged that the heart can be classified in either way.

It might be useful to look upon complete transposition complexes as having multiple variables producing many combinations. The variables are 1) pulmonary stenosis or

atnesia, or hypoplasia of the aorta with or without coarctation, 2) complete, partial or no ventricular septation, and 3) normal

atrioventricular orifices or mitral or

tricus-pid stenosis or atresia, or common

atnio-ventricular orifice.

From the surgical point of view, the

above classification of complete tnanspo-sition has its place in deciding whether a lesion is amenable to surgery, and the type of surgery to be employed. In complete transposition with normal architecture, with or without ventricular septal defect or with

mild pulmonary stenosis, partial correction by transplantation of the inferior vena cava and night pulmonary veins may be under-taken. In complete transposition with non-mal architecture with marked pulmonary stenosis, and other types of complete trans-position with pulmonary stenosis or atnesia, an aortico-pulmonany,

subclavian-pulmo-nary, or superior vena cava-pulmonany

artery anastomosis may be performed. Complete transposition with abnormal

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atnesia or with common atnioventnicular

on-fice are characteristically associated with pulmonic stenosis and atresia, while com-plete transposition with tricuspid stenosis on atresia, on with common ventricle, or single ventricle and small outlet chamber are more

commonly associated with increased pul-monary flow. The two complexes, complete transposition with common ventricle and that with single ventricle and small outlet chamber, are very closely related

pathologi-cally.

REFERENCES

1. Lev, M., and Saphir, 0. : Transposition of the

large vessels. J. Tech. Methods, 17:126, 1937.

2. Lev, M., and Saphir, 0. : A theory of

trans-position of the arterial trunks based on the

phylogenetic and ontogenetic development

of the heart. Arch. Path., 39: 172, 1945.

3. Lev, M. : The pathologic anatomy of cardiac

complexes associated with transposition of

arterial trunks. Lab. Invest., 2:296, 1953.

4. Lev, M. : Congenital heart disease, in A Text

on Systemic Pathology, Vol. 1, edited by 0.

position den Herzostien-ein Versuch der

Erkl#{228}rung dieser Erscheinung: Die

Phorono-mie den Herzentwicklung als morphogene-tische Grundlage den Erkl#{228}rung; die

Pho-ronomie den Herzentwicklung. Z. Anat.

Ent-wicklungsgesch., 100:563, 1933.

10. Taussig, H. B.: Complete transposition of the

great vessels : clinical and pathologic fea.

tures. Amen. Heart J., 16:728, 1938.

11. Harris, J. S., and Farber, S.: Transposition of

the great cardiac vessels, with special

refer-ence to the phylogenetic theory of Spitzer.

Arch. Path., 28:427, 1939.

12. Becker, M. C., and Bnill, R. M. : Complete

transposition of great vessels : report of three

cases and review of the literature. Arch.

Pediat., 6,5:249, 1948.

13. Edwards, J. E. : Congenital malformations of

the heart and great vessels, in Pathology of

the Heart, Ed. 2, edited by S. E. Gould.

Springfield, Illinois, Thomas, 1960, p. 260.

14. Taussing, H. B.: Congenital Malformations of

the Heart. New York, The Commonwealth

Fund, 1947.

15. Lev, M., and Kaveggia, E. : The etiology and

pathogenesis of congenital heart disease, in

Cardiology, edited by A. Luisada. New York,

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1961;28;293

Pediatrics

Maurice Lev, Victor M. Alcalde and Thomas G. Baffes

ARTERIAL TRUNKS