Anomalous Pulmonary Venous Return - Circulation

Anomalous Pulmonary Venous Return 

HU A. BLAKE, COL., ROBERT J. HALL, LT. COL. and WILLIAM C. MANION 

Circulation. 1965;32:406-414 

doi: 10.1161/01.CIR.32.3.406 

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Anomalous Pulmonary Venous Return 

By Hu A. BLAKE, COL., MC, ROBERT J. HALL, LT. COL., MC, 

AND WILLIAM C. MANION, M.D. 

M ANY detailed studies concerning isolated 

aspects of anomalous pulmonary 

venous return are available. Surveys encompassing 

the entire spectrum of such anomalies 

are, however, generally lacking. For purposes 

of this presentation, 113 selected instances of 

anomalous pulmonary venous return encountered 

at Brooke General Hospital, Walter 

Reed General Hospital, and the Registry of 

Cardiovascular Pathology of the Armed 

Forces Institute of Pathology have been reviewed 

and correlated with related material 

from the literature. To obtain completeness, 

tempered with brevity, the better known 

anomalies, while mentioned, are discussed 

only briefly, whereas the less well described 

transitional forms are discussed in detail. 

Highly schematic diagrams are used to portray 

some 27 variations in this theme. It is 

hoped the wide anatomic spectrum shown 

will promote understanding and interest. 

Figure IA illustrates the most frequently 

described variety of anomalous pulmonary 

venous return in which all or a part of the 

veins from the right lung drain directly into 

the right atrium. In two of 26 such instances 

the septum interestingly was intact. It should 

be emphasized that a normal connection does 

not always mean normal drainage. We, like 

others, have seen examples in which indicatordilution 

curves indicated normal drainage and 

at surgery the vein connected anomalously." 2 

Figure iB, a situation encountered twice, 

we think important as it represents a paradox 

not easily explainable embryologically. Entry 

of the right pulmonary veins into the left 

atrium and entry of the left pulmonary veins 

into the right atrium seemingly defy ex- 

From Brooke General Hospital, Fort Sam Houston, 

Texas; Walter Reed General Hospital, Washington; 

and the Registry of Cardiovascular Pathology, Armed 

Forces Institute of Pathology, Washington, D. C. 

406 

planation on the basis of simple reabsorption 

of the common pulmonary vein followed 

by abnormal septation. We are not familiar 

with others encountering this anomaly. 

Eight instances of total anomalous pulmonary 

venous return to the right atrium 

(fig. IC) were seen. When there was a large 

defect lacking a septal ridge posteriorly, it 

was often difficult spatially to orient the 

venous orifices. The incidence of this anomaly 

might have been increased, if one had been 

less critical in this regard. 

Figure ID differs only in the addition of a 

left superior vena cava. The emptying of this 

vessel into the coronary sinus distinguishes 

it from a persistent left innominate vein.3 

Figure IE is a case associated with four 

venae cavae. The flow through the persistent 

left inferior vena cava usually represents hepatic 

vein drainage. This is one of two cases 

we have personally encountered with all four 

venae cavae persisting. (See also figure 6, 

where there are two inferior venae cavae.) 

We have observed seven methods whereby 

anomalous pulmonary venous return has occurred 

at the supracardiac level. These were 

(1) partial or (2) complete anomalous pulmonary 

venous return directly into the right 

superior vena cava, (3) partial return from 

the right through the azygos vein, (4) partial 

from both lungs through the azygos vein, (5) 

partial return via a left innominate vein, (6) 

complete return via a left innominate vein, 

and (7) partial return from both lungs via a 

levo-atrial cardinal vein to the right superior 

vena cava. In the interest of continuity, four 

of these seven variations will be covered now 

with discussion of the remaining three 

more appropriately being placed later. 

Figure IF depicts the findings in twenty 

cases of partial or total right pulmonary 

venous return into the right superior vena 

cava. Embryologically, these defects are often 


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Circulation, Volume XXXII, September 1965

ANOMALOUS PULMONARY VENOUS RETURN 

Figure 1 

Schematic representation of types of anomalous venous return. 

associated with high, sinus venosus-type atrial 

septal defects.4 5 Such high defects were observed 

in 17 instances, typical secundum defects 

occurred twice, and in one instance the 

atrial septum was intact. 

All pulmonary veins entering the right 

superior vena cava to date have done so at 

or below the azygos entry. This finding is 

interesting in view of the frequency of anomalous 

pulmonary venous return on the left 

finding its way to the left jugular-subclavian 

juncture. Higher, more peripheral connections 

suspected on catheterization, but not proved 

at surgery, are explained by the perfused 

unexercised right arm giving unusually high 

Circulation, Volume XXXII, September 1965 

407 

oxygen-saturation values. Entry at or below 

the azygos level enhances surgical repair. 

Complete anomalous pulmonary venous return 

to the right superior vena cava, found 

seven times, is depicted in figure 1G. In this 

small group, three anatomic features were 

apparent. First, the lowermost end of the 

cava was consistently dilated. Secondly, the 

anomalous orifice was usually quite large, 

placed directly posterior, and single. Thirdly, 

the accompanying septal defect was highly 

placed. These factors again favor surgical 

repair. 

Anomalous pulmonary venous return 

through the azygos vein may exist in part or 



408 

in whole. Figure IH depicts partial return of 

all the right pulmonary veins to the right 

superior vena cava through the azygos. One 

might also suspect an absence of the suprarenal 

portion of the inferior vena cava to accompany 

this defect.Y This was not true in this 

case. 

Total anomalous pulmonary venous return 

to the superior vena cava through the azygos 

and persistent left innominate veins was seen 

but once and is depicted in figure 1I. The 

term persistent left innominate vein is preferable 

to persistent left superior vena cava when 

this remnant of the left anterior cardinal vein 

fails to maintain a connection with the coronary 

sinus or left atrium. 

With these four descriptions completed, the 

three remaining variations of anomalous pulmonary 

venous return to the right superior 

vena cava will be inserted at points considered 

more advantageous in regard to continuity. 

Low cardiac and immediately infracardiac 

connections are the next two groups to be 

considered. In these categories, five interesting 

and often extremely complex connections 

were encountered. These were (1) partial to 

the inferior vena cava, (2) partial and (3) 

complete to the coronary sinus, and (4) 

partial and (5) complete to the portal 

system. 

First discussed are partial connections 

from the right lung to the inferior vena cava. 

The seven instances encountered have proved 

interesting in that they are somewhat in 

variance with published data. In five of them, 

there was an associated atrial septal defect, 

in two there were none. Additionally, both 

supradiaphragmatic and infradiaphragmatic 

inferior vena cava connections were seen. The 

anatomic configuration of these channels has 

been studied in detail, as correction usually 

entails transplantation to a higher level. In 

two of the four cases suitable for study, approximately 

one third of the circumference of 

the anomalous channel was visible superficially 

as it lay grooving the lung parenchyma. 

From the embedded portion of the circumference, 

small pulmonary venous branches 

serially joined the main trunk in a manner 

BLAKE ET AL. 

analogous to the coniferous branching of the 

spruce. In the remaining two cases, a large 

dichotomous branching took place immediately 

prior to this channel joining the inferior 

vena cava. This form of branching, as opposed 

to the smaller serially branching type, 

introduces the danger of a lower lobe venous 

infarction should either of these large limbs 

be divided to permit mobilization and higher 

reimplantation of the anomalously entering 

pulmonary vein. 

Figure 2A demonstrates the partial supradiaphragmatic 

drainage of the right lung 

into the inferior vena cava encountered six 

times. Drainage of blood from the left lung 

into the inferior vena cava is apparently rare. 

D'Cruz has reported a recent case.7 Interestingly, 

in our small group, only two cases 

demonstrated the features of the vena cavabronchovascular 

syndrome described by 

others.8-1 Likewise, the value of "commalike," 

"sickle-shaped," "scimitar-shaped" paracardiac 

shadows in the lower right lung 

field as a pathognomonic indication of anomalous 

pulmonary venous return into the 

inferior vena cava should probably be deemphasized. 

Figure 3 shows such a paracardiac 

shadow, which though it was a long 

redundant pulmonary vein emptied normally 

into the left atrium.12 Pulmonary veins of 

this size with elongation, redundancy, and a 

normal left atrial point of entry probably best 

fit the category of "varicosities" of the pulmonary 

vein.13 

Figure 2B demonstrates the singly encountered 

partial drainage of the right lung 

into the inferior vena cava below the 

diaphragm. As the diaphragm was not opened 

at surgery, the anomalous pulmonary venous 

channel's course after piercing the 

central leaf of the diaphragm is obscure with 

but catheterization evidence of its high entry 

into the inferior vena cava. Cooke has described 

a somewhat similar case.14 Total infradiaphragmatic 

pulmonary venous connections to 

the inferior vena cava have been reported and 

twice successfully repaired.'5' 16 One should 

distinguish this form prognostically from 

infradiaphragmatic drainage into the portal 





Figure 2 

Schematic representation of types of anomalous pulmonary venous return-continued. 

system, as the physiologic disturbance produced 

by the latter is infinitely greater. 

Semantically representing low cardiac, 

rather than infracardiac drainage, anomalous 

return into the coronary sinus varies from one 

vein to that of all the veins returning by this 

route. Of the former, a single case, illustrated 

in figure 2C, was encountered. Bilateral lower 

lobe drainage into the coronary sinus has 

occurred.17 It should be noted that infrequently 

the coronary sinus fails to drain into the 

right atrium. In atresia of the orifice of the 

coronary sinus, the coronary sinus drains 

retrogradely into the left atrium through a 

persistent Bochdalek's foramen.1820 Figures 


409 

4F and G later illustrate forms of this anomaly. 

Figure 2D represents a case of total coronary 

sinus drainage. The anatomic proximity of 

the enlarged orifice of the coronary sinus 

to the tricuspid valve presumably encourages 

streamlining of the coronary sinus-pulmonary 

venous return. This may result in 

the catheterization finding of higher oxygen 

saturations in the right ventricle and pulmonary 

artery than in the left atrium, left ventricle 

and the aorta, a point of diagnostic importance. 

Repair of such an anomaly is shown in 

figure 5. The narrow intervening bridge of 

tissue lying between the atrial septal defect 



410 

Figllre 3 

Arrows identify a vertically coursint, right paracardiac 

ptulmonary veffi. After a circaCtitons totc, this 

vein entered the lcft atritum iiormailh 

and the enlarged coronary siIIus orifice is excised 

and the two then confluent openings 

are roofed over diverting the pulmonary venouis 

and the coronary sinus return (the volume of 

the latter's desaturated blood is considered 

to be inconsequential) into the left atrium. 

Production of surgical heart block is to be 

avoided.21 

A variant of coronary sinus drainage is 

depicted in figure 2E. It has been inferred 

that mixed levels of anomalous pUlmonary 

venous drainage do not occur or are quite 

rare. Gott et al. reporting 30 cases of total 

anomalous pulmonary venous drainage stated, 

"Of the cases cited in the literature as having 

drainage via the left superior vena cava, 

none appears to drain in this fashion-communicating 

proximally with the coronary 

sinuis and distally with the innominate 

vein."22 Similarly, Darling's group in a review 

of 17 personal cases and a survey of 

the available literature (80 cases) could find 

but a single case of mixed levels of drainage.28 

Interestingly, nine instances of suich double 

0BLAKE ET AL. 

levels of drainage as illuistrated were encounitered 

in this series. 

In a single additional case, the left upper 

lobe vein drained normally inito the left 

atriu-m as shown in figu-re 2F. 

A transitional form leading tovards the 

classic "figure-of-eight" anomaly is illuistrated 

in figuire 2G. In this, and the followiing 

anomaly, the clinical anid radiologic patterni 

can apparently be inodified considerably, 

depending uipon xx hetlher or inot the perIsistent 

left eaval remnant passes as it uisuially does 

anterior to the puilmonary artery or posteriorly 

as is occasionally seen. In passing posterior 

to the pulmonary artery, the anomalous channiel 

may become obstructed with impeded 

venous ouitflow from the lulngs simullatinig an 

infradiaphragmatic type connection.24 

Figure 2H represenits the classic form of 

total anomalous puilmonary venouis return. 

Diagnostic-wise, xwide variatiois have occurred 

in the time of appearance of the 

classic chest roentgen ograin. The chest roentgenogram 

of a 3-montlh-old inifant xxas most 

suggestive, wvhereas in a 13-year-old the 

configuiration vas niot particuilarly impressive. 

The monotonouisly equlal, markedly elevated 

oxygen-saturations throughotut all chambers, 

pulmonary and peripheral arteries is highly 

suggestive of total anomalouis pulmonary vein 

drainage, especially of the supracardiac type. 

The values found in one such case are shown. 

Twvo associated anomalies have greatly 

inflicenced the cliinical courses followed by 

these patients. We have repaired suich anl 

anomaly in an older child in xvhich there 

was only the slightest clinical disability. This 

clinaical well-being apparently resulted from 

the coexistence of a mild pulmonary stenosis 

which beneficently prevented pLilmonary 

flooding and encoturaged right-to-left shunting 

through a large atrial septal defect. At 

the other extreme, marked early deterioration 

has been seen in quite young infants when 

the atrial commiunication was small, allowing 

little blood to reach the left side of the 

heart. In a 9'2 pound infant recently operated 

upon the diameter of this commniunication 



Cic ulafton, Volume XXXI!, September 1965


Figure 4 

Schematic representation of types of anomalous pulmonary venous return-continued. 

(patent foramen ovale) was a mere 5 mm. 

In figure 2I an anomaly encountered three 

times is shown. Should disease or removal of 

the right lung occur, survival would be possible 

only in the presence of an atrial septal 

defect.25 The variant encountered once, seen 

in figure 4A, does not, of course, carry these 

dire implications. 

In figure 4B a transitional form combining 

supracardiac and infradiaphragmatic portal 

drainage is illustrated. The heart and lungs 

showed no abnormality other than the anomalous 

pulmonary venous drainage and the 

atrial septal defect. This technically correctable 

association of anomalies, with the lack of 


411 

a severe intracardiac malformation, invites 

staged surgical repair. 

Total infradiaphragmatic pulmonary venous 

drainage, as illustrated in figure 4C, was 

encountered twice. The pulmonary veins 

formed a common trunk which passed 

through the esophageal hiatus to enter the 

portal vein by way of an enlarged gastric 

(coronary) vein. The clinical and radiologic 

picture is that of obstructed pulmonary venous 

outflow.26' 27 The better prognosis of an 

infradiaphragmatic drainage emptying into 

the inferior vena cava or the hepatic veins 

has already been mentioned. 

Figure 4D, an example of mixed levels of 



412 

mnterotrol septol 4 

defect- 

EnIcHroed coronory25 

snus or fic 

BLAKE ET AL. 

Figure 5 

l1otal aitoinaloits pultnonary viCUoU.s drainag,e inlto the coronIarIy sinus. Isn (1) the intraotrial 

anatomIy is showtn. In (2) the thin1 scpjttln of tissue cdivicvidig the coronan,J sinu2Xs and the atrial 

septal dlefcet is beinig dlivided. In (3) the resultanit confluent complex hZas beent roofed over 

diverting the pulmonary venouis anid coronary sinuis return in1to tte left atrium???. 

drainage, differs only in the presence of a 

left upper lobe vein drainiing normally. Figure 

6 is nearly identical except that there are 

two inferior venae cavae present. 

The anomaly depicted in figure 4E is interesting 

embryologically. The heavily dotted 

circle diagrammatically represents a thick 

muscular diverticulum found growing outward 

from the back wall of the left atrium. 

Had this diverticulum succeeded in connecting 

xvith the common pulmonary venious 

trunk, tvo possibilities can be envisioned. If 

the connection were sufficiently large, the 

infradiaphragmatic channel vould likely disappear. 

lf the connection were a small or 

constricted one, a cor triatriatum might have 

resulted upon involution of the portal connection. 

The next three figures (4F, G, and H) 

represent left superior venae cavae emptying 

directly into the left atrium. In many hearts, 

immediately above the mitral mural lcaflet, 

a large thebesian vein orifice is seen, xhich 

Bochdalek, Lancger, and Tandler have noted 

as an almost conistant feature in the left 

atrium. In atresia of its ostium, the coroniary 

sinus generally drains retrogradely to enter 

the left atritum at this point (fig. 7) .lS-20 

A left superior vena cava directly entering 

the left atriu-m also does so by this venous 

orifice. 

The patient represented by figure 5F had 

atresia of the coronary sinus orifice, a primnum 

atrial septal defect, and a left superior vena 

cava (receiving oine ainomalous pulmnonary 

vein) enterinig the left atriLim directly. 

In figture 40 a tiny 2-mm. slit marked the 

hypoplastic coronary sinus orifice. Atresia 

of the mid-portion of the sinus is postulated 

with retrograde flow into the left atrium 

throuigh the orifice also discharging blood 

from the persistent left suiperior venia cava. 

The correctness of this assumption, hovever, 

could not be fully ascertained by 

inrulatOion, Volume XXXI!, September 1965 




Figure 6 Figure 7 

For cllaiity, tlhe headt excepting a 

) t posterior shell of the 

ft~~~~~~~~~~~~rrot; points to th-e sJco)r2darily eiilaigetl ot-ifice of the 

essentially common atriulm htas been removed. Three vein of Bochdalek, the primary cause of this enlarge 

cavae-a ,'ight superior, and a rig/it and left inferor ment being an atresia of the coronanjy sintus orifice. 

enter th/c atria. Transparency of the liver and atria Taken front Bredt's figure 16.18 Bredt, II.: Formdeupermits 

visualization of the posterior-lying common tung und Eristehung des missgebildeten iensc/dichen 

pulmonary veniouis trunk's course and connection with Herzens. Virchow Archiv fur Pathologische Anatomie 

e c 

the cor-orlary veinr lAon- the lessert gasti-ic ctirvati-1re. und Physiologie, Bd. 296, S. 114 (1936) Springer-Verlag, 

Berlin-GC ttim'gen-Hteidel/erg. Reproduced by pervisualization 

and bidirectional probling. mnission of t1/e publishers. 

Cor triatriatum is usually an abnormality 

of the size, not the site, of the left atrial 

puLlmonary venous connection. In figure 4H 

a cor triatriatum is combined with the 

presence of a "levo-atrial cardinal vein" as 

described by Edwards.28 Because of the 

small commoni pulmonary veini-left atrial 

communication, a venous connection between 

the mid and the anterior cardinal systems 

persisted. As the major pulmonary venous 

flox was directed into the right superior vena 

cava, the case hemodynamically represents 

one of anomalous puilmonary venous return 

and rnot one of cor triatriatum, thus accounting 

for its inclusion in this presentation. 

Summary 

One hundred and thirteen selected anoma- 

Clircwlation. Vol/nir XXXII. Scpenrnber 1965 

413 

lous pulmonary venous returns have been xvell 

documented anatomically. Twenty-seven variations 

were seen. With the use of simplified 

diagrams, these have been correlated xvith 

related cases in the literature. Emphasis has 

been placed on transitional forms with one 

anomaly shading off into the next, rather than 

upon a series of neat, compartmentalized, 

classic types of aniomalous pulmonary venous 

return. It is hoped that the broad anatomic 

spectrum showvn xvill promote additional 

understanding and interest. 

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414 

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Anomalous Pulmonary Venous Return - Circulation

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