VOL.
4
No.
225,
THE
VARIOUS
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REPORT
OF
YORAM ALBERT
By
FORMS
THREE
NEW
BEN-MENACHEM, N. BREST, M.D.,!I
0.
OF
CASES
PULMONARY
AND
REVIEW
M.D., t KOSON KURODA, PRESTON COPELAND,
HOUSTON,
TEXAS
AND
VARICES* OF
M.D.,
E. and
M.D.,t
PHILADELPHIA,
THE
LITERATURE
ROSS
KYGER,
JOHN
III,
D. COAN,
M.D.,
M.D.t
PENNSYLVANIA
ABSTRACT:
True
pulmonary
vanices
are
congenital
local
dilatations
veins, with normal or collateral transpulmonary Pulmonary vanices do not cause pulmonary varices can further become dilated by pulmonary valve disease. Embryologically, venous components an individual unobstructed True
over
pulmonary transpulmonary
pulmonary
the
which collateral
do
usually,
not
only
42
occurred drainage
the in
flew2-4’8-’O’’2’’5-_’7’’9-21’22’23’26’
the
monary
varices.
two
main
REPORT
categories
OF
of
or
hypertension
due
a time into the
primitive system,
that left do
not
enabled atrium. change
to mitral splanchnic atresia
or
adoption in
of of
diameter
treatment.
pattern.
During
dilated,
tortuous
the right the right tered the visualization
and three doubtful2”4 cases were published. Review of the literature revealed differences in the pattern of the pulmonary vanices in several cases, possibly reflecting basic differences in the formation of varices in the embryo. The purpose of this paper is to present three cases and discuss the differences
vein
atrium. but existing
residual venous
symptoms,
require
29-35,38-40
between
at
a pulmonary
into the left hypertension,
venous
may represent the pulmonary
produce
do not
varices are rare. Since of a patient by Hedingen
report
190718
and,
varices into
vein
varices
years,
P ULMONARY first
pulmonary incorporated
of
drainage venous
the
venous
pulmonary
phase
veins
(Fig.
were
2B),
seen
in
lower lobe; these veins drained into superior pulmonary vein, which enleft atrium normally. There was no of a right inferior pulmonary vein.
pul-
CASES
CASE I. H.W., a 50 year old female, was referred for a routine examination because of pulmonary tuberculosis in a member of her family. A chest noentgenogram (Fig. i) revealed dilated vascular
structures
in the
right
lower
lung
field
medially. Review of previous roen tgenograms of the chest showed that these vascular shadows have existed, unchanged, for the past 24 years. The patient was asymptomatic and there was
no evidence ease. evidence
pertension. angiogram *
of congenital
Pulmonary
From
of Radiology,
of
or acquired
angiography pulmonary
The arterial (Fig. 2A) the Departments and
Medicine,
arterial
phase revealed
not
or
venous
disreveal
hy-
FIG.
of the pulmonary a normal atrial
of Radiology,t Thomas
heart
did
i.
Case
vascular field and
i. Chest roentgenogram. markings in the right lower right hilum.
and Surgery,* The University of Texas Medical
Jefferson
University
Hospital,
88i
Philadelphia,
School
Pennsylvania.
at Houston,
and
Prominent medial lung
the
Departments
Ben-Menachem
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882
FIG.
2.
Case
the right
Selective
right
pulmonary
superior
pulmonary
pulmonary
vein.
old female,
II.
R.W.,
a 53 year
clinical
suspicion
space-occupying
lesion.
revealed
aneurysm
carotid (Fig. , lobulated, Pulmonary
a berry
of
an
Cerebral of
right
was
arterial
During the pulmonary
arterial arteries
Case
II.
or
phase were
venous
the
angiography the
left
internal
Note
-
lobulated
into
monary
vein
atrium. CASE
III.
Angiography grams
in the soft
tissue
phase
the
which
phase
drain
E.V.,
a 62
showed 6,
A
(A) posteroanterior mass in the right
were
varicose, side
evaluation
5, A and
lung
drained
left
parathyroid
(Fig.
arterial
varices
(Fig.
right
a single, The
for
The
1975
into
vein.
from
draining
inferior
position. (A) lower pulmonary
the venous
mitted
hypertension.
of the chest, round,
c
veins
left
(Fig. 4, A and B), the found to be normal.
Roentgenograms
anterior
During
ad-
intracranial
artery. Roentgenograms of the chest A and B) revealed a rounded, slightly soft tissue density in the right hilum. angiography showed no evidence of
pulmonary
FIG. 3.
angiogram,
DECEMBER,
(B) During the venous phase, large, There is no right inferior pulmonary
arteries.
for
CASE mitted
i.
normal
shows
et al.
normally
was year
of
common
all
B),
found
to
be
pulinto the
normal.
old
female,
was
ad-
the
left
hyperparathyroidism.
an gland. and B)
and (B) hilum.
adenoma Chest
revealed
lateral
of
roentgeno-
a round,
projections.
VOL.
No.
125,
The
4
Various
Forms
of Pulmonary
883
Varices
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V
4. Case
FIG.
II.
(A) Midstream angiogram,
anteroposterior arterial
and (B)
phase.
Note
slightly lobulated, soft tissue density in the right hilum. Pulmonary angiography showed no evidence of arterial or venous pulmonary hypertension. During the arterial phase (Fig. 7, A and
B)
normal
arterial
architecture
was
found. B), the
During the venous phase (Fig. 8, A and veins of the right lower and middle lobes were found to be draining into a varicose, right inferior pulmonary vein which drained normally into the left atrium. The right superior pulmo-
nary vein normal.
and
both
left
pulmonary
veins
were
varix ment the
anterior oblique selective pulmonary arteries.
normally
the
draining
absence
tension
due
of by
According
to
of
pulmonary
a segenters but
veins;’0
venous
disease,
stability
the over
in
hyperlesions
many
are years
9,12,14,16,17,19,29,32
the
this
7
definition,
cases as well
at
hitherto as our
least
pub-
own
Cases
and II, are not true varices, as they all show anomalies in the involved pulmonary veins, even though the final drainage is into the left atrium.
i
DEFINITION
traditional
pulmonary
pulmonary
to heart
characterized of observation.”
seven
right
is a localized enlargement of of a pulmonary vein which left atrium normally,2 or prominent,
lished,2’4’20’23’26’31’9 DISCUSSION
The
right normal
definition
of a pulmonary
r
r FIG.
.
Case
oblique
ii.
Venous
projections.
phase
Large
pulmonary hilar varices
of
angiogram drain the
in the (A) anteroposterior entire right lung into the left
and atrium
____________________ (B)
right anterior superiorly.
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884
ETIOLOGY
It is generally agreed that pulmonary varices are local congenital anomalies without significant relation to other existing congenital cardiac, pulmonary, or other disorders, and without known etiology.2”30’32’34 The development of the pulmonary venous system and its anomalies has been extensively studied.”6’24’25’28’42 Most probably the defect occurs at the time of transition of the lung from splanchnic venous drainage to pulmonary venous drainage of
et al.
Ben-Menachem
proper,
the
embryonic
ence of tributed these
during
horizons
development.
local varicose to weakness
segments.’
veins
pulmonary as a possible
venous cause
in
the
system of such
is, however, no evidence walls of the splanchnic It is hard basis of
than
The
segments of the venous
Incorporation
splanchnic
weaker
xIIi-xvI28
those
to explain differences
of primitive
newly
developing
was suggested weakness;2 there
to suggest veins are
of the
atin
that the basically
pulmonary
the dilatation in wall structure.
veins. on
the In
cases, in which the of the varicosities
layers,
including
found cases
the
1975
microscopic strucwas studied, all elastic
fibers,
were
be normal.9”8’22’27’29 Only two reported as showing an abnormality: in one,3’ the wall was thinner than normal and the muscular layer was replaced by fibrous tissue; in the other,32 the wall was very thick and fibrous.
to were
DISTRIBUTION
Sex the
existwas walls
most ture
DECEMBER,
and
Age.
literature
three
Among cases reported in females, 19 males, and sex was not reported),2’4”5 (28
whose
the
varices
were
32.2
years
mean
in
years In
mean a small
in the series
females. such as
ences
in age
and
first
sex
discovered
the
one,
are
most as the
age
37.5 differ-
cannot
is usually
an incidental
finding
tigations
for
unrelated
conditions.
Localization. varices were
In found
most in the
and
this
distribution
regarded as significant and ably coincidental, particularly
at
males
during
be probvarix inves-
reported cases, right lower and
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VOL.
No.
225,
middle lobe tional hilar vein lung.
right
not
understood
upon
upper
in this
cases monary THE
did
11.
VARIOUS
True majority
patients),
lobe
and
and
will
paper.
In
have
PATTERNS
DILATED
OF
Forms
of Pulmonary
Varices
885
left upper Ten addias having as to the an entire sparing of
lower
lobe
is
be speculated of the
reported
bilateral
PULMONARY
PULMONARY
Pulmonary of the and
left
not
none
the patient varices.
AND
30
Various
lobe (20 patients) or the and lingula (i. patients). patients were reported varices, without specification involved, or varicosities over The reason for the relative
the
the
The
4
pul-
VARICES VEINS
Varices. These are reported cases (about our
Case
III
belongs
in
this group. The only anomaly is a locally dilated pulmonary vein or veins in part of a lung or its hilum. Two pulmonary veins exist on each side and all enter the left atrium normally. We shall assume that this anomaly may be the result of incorporation of primitive splanchnic venous segments in the newly developing pulmonary venous system, as proposed by Bartram and Strickland.2
B. Dilated Transpulmonary 1”enous Collaterals. Seven patients2’4’”31’39 and our Cases I and ii are recorded as having a single right or left upper or lower pulmonary vein, with dilated, tortuous (varicose) veins draining the rest of the lung into them. The single pulmonary vein enters the left atrium normally. This type of anomaly can theoretically be created by secondary atresia of the pulmonary vein after the separation of the splanchnic venous drainage, but before lobar separation of the pulmonary veins was completed, thereby allowing transpulmonary collaterals to develop. Had the atresia occurred earlier, pulmonary-splanchnic drainage would have been permanently established; a later occurrence would have resulted in individual pulmonary vein stenosis with all its symptomatology and sequelae.5’24’25’36’37 C. Dilated Pulmonaty 1eins with Abnormal Insertion into the Left Atrium. Two
FIG.
7. Case gram, arterial right anterior nary arteries.
Selective
In.
right
pulmonary
angio_ and (B)
(A) Anteroposterior projections. Normal
phase. oblique
pulmo-
cases are reported, one with a left superior pulmonary vein26 and one with a left common pulmonary vein,20 inserting too far to the
right
veins
as true
into
were
the
dilated
varices.
left they
atrium. cannot
Although be
regarded
the
Ben-Menachem
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886
et al.
2975
DECEMBER,
varices. Whether the dilated veins were true varices or not is difficult to say on the basis of the published material; and in the presence ofmitral stenosis, their bilaterality and hilar location suggest that they were probably chronically distended main pulmonary veins rather than true varices. Bryk7 describes seven patients with mitral insufficiency, and with markedly dilated right pulmonary veins. The localization of the dilatation in the right veins in Bryk’s cases can be explained by the direction of the regurgitation flow, which would be preferentially into the right pulmonary veins. These, also, are not true pulmonary varices. SYMPTOMS
When pulmonary varices are the patient’s only abnormality, they do not produce symptoms.2’3”3 In most reported cases the discovery of the varix was on routine chest roentgenograms or during investigation of cardiac or other diseases. Hemoptysis was reported in five patients.2”8’27’3’
In
one”
it
originated
from
bronchiectasis; in another27 bleeding may have been due to pulmonary tuberculosis. Three 1829 presented with cough, which cannot be shown as being directly
8. Case
FIG.
gram,
in
anterior
pulmonary
In.
the
Venous phase of pulmonary angioanteroposterior and (B) right
(A)
oblique vein
into
the left atrium.
vein
is normal.
projections. is varicose,
The
The right but drains
right
superior
inferior normally
caused
by
the
with
congestive
causes,
pulmonary
THE
but
heart unrelated
RELATIONSHIP
“Pulmonary
Varices”
described mitral
a patient stenosis and
Viamonte
in Mitral and
StenoLePage4’
(their Case II) with bilateral pulmonary
of
OR
Most alies disease:
commonly, were
pul-
ciency;8”9’20’21’33
patients three
various
varices. ACCOMPANYING
DISEASES
AND
VARICES
accompanying
congenital five
of
the
BETWEEN
PULMONARY
D.
failure to
ANOMALIES
sis andlnsufficiency.
presence
monary varices. One patient23 presented with a middle lobe syndrome, directly related to pressure on the middle lobe bronchus by the varices. Two patients presented with dysphagia,20’26 due to pressure on the esophagus by a large left pulmonary vein which inserted abnormally to the right of the left atrium. Both these patients are not regarded as having true varices. Most symptomatic patients presented
anom-
or
acquired
heart
had
mitral
insuffi-
had
mitral
steno-
VOL.
225,
No.
The
4
Various
Forms
of Pulmonary
and one each had coarctation of the aorta,’5 aortic stenosis and pseudocoarctation,2 common origin of both great vessels from the right ventricle,’8 ventricular septal defect and patent ductus arteriosus,4#{176} malrotation of the cardiac chambers,2 and absence of the right hemipericardium.3#{176} Other associations included pulmonary tuberculosis,27 systemic hypertension,22 left upper lobe pulmonary emphysema,2 intracranial aneurysm (our Case ii), and hyperparathyroidism (our Case III). Three patients had pulmonary arterial anomalies, including absence of the right main pulmonary artery,’2 hypoplasia of the descending branch of the right pulmonary artery,” and a trifurcation, rather than bifurcation, of the right pulmonary artery.3 Two patients2 had partial anomabus venous drainage contralateral to the side of the pulmonary varices. Varices are not created by heart disease, and their existence in cardiac patients is purely coincidental.2” In patients with pulmonary venous hypertension due to mitral stenosis or insufficiency, pre-existing varices may become wider.” In such cases, progression of the congestive heart failure may make the presence of an enlarging varix rather threatening,’ by creating conditions for rupture. In one patient with mitral insufficiency’9 the varices became progressively distended over a seven year period and disappeared following surgery. In another,2#{176} dysphagia due to a distended left pulmonary vein with an aberrant insertion into the left atrium and pressure on the esophagus disappeared after treatment of the patient’s congestive heart failure.
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sis;8.9.16
COMPLICATIONS
Two patients29”2 suffered a cerebral which was possibly Hedinger’8 felt odenal ulcer was from
deaths from
DEATH
are reported to have embolus, the source of in the varices. that his patient’s ducaused by an embolus
a pulmonary
Three sulted
AND
reported ruptured
as having pulmonary
re-
887
varix.’”2 In one, the varix grew rapidly with aggravation of existing right heart failure. In another, rupture may have been caused by erosion of the varix by pulmonary tuberculosis in the adjacent lung tissue. The complete clinical picture of the third patient’2 is not available. A fourth patient29 is reported to have died of a cerebral embolus, but the description of the clinical picture in this patient is strongly suggestive of congestive heart failure. ROENTGENOGRAPHIC
DIAGNOSIS
DIFFERENTIAL
AND
DIAGNOSIS
The smooth, rounded or lobulated soft tissue density of the varix on the plain chest roentgenogram and tomogram should be differentiated from other pulmonary or hilar mass lesions, including those that are neoplastic, granulomatous, lymphatic, or vascular in origin.2” 8,10, 1316,19.20,21,38 At least one patient’4 was given treatment for two years for pulmonary tuberculosis, until the true nature of the mass on the chest roentgenogram was revealed. A most important differential diagnosis is that of pulmonary arteriovenous 1330 which may have an identical roentgenographic appearance. Partial anomalous pulmonary venous return should also be included in the differential diagnosis.’9 The Muller maneuver enlarges the size of pulmonary varices, and the Valsalva maneuver decreases it in many, but not all patients.2”6”9 The only safe criterion for the diagnosis of pulmonary varices is the opacification of the varices during the venous phase of pulmonary angiography. Demonstration
of
normal
pulmonary
ar-
terial and venous pressures is necessary to exclude heart disease as an etiologic factor. An important additional criterion is the non-growth
of
pulmonary
varices
over
long periods of follow-up. Eleven tients3’9”2”4”6”7”9’29”2 and our Case follow-up chest roentgenograms over ods
varix.
are a
Varices
ranging
from
two
any appreciable change pulmonary varices.
to
24 years,
in
the
I
pahad peri-
without
size
of
the
et al.
Ben-Menachem
888
the
TREATMENT
Treatment
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matic
is not
patients
disease.’4’21”#{176}’3’ failure, ces, the
necessary
who
who
with
heart
of the
reduce of
14.
heart
right
enlargement
can be expected to lesions with treatment
tive
of
the size of their conges-
15.
hemoptysis
as
in
exists
pulmonary
the
and
patient
in
is directly
varices,
related
should
be 17.
considered.2 Yoram Department
of
Hermann
Ross
3203
Radiology
Houston,
I 8.
Sterling
AUER,
77025
19.
2.
J.
of
variations.
Jo!, BARTRAM,
D.,
7. Canad. 4.
human
20.
pulmonary
Anat.
Rec.,
BENFIELD,
A.
Radiologists,
J. R.,
GoTs,
Anomalous single ing parenchymal
1972,
R. E.,
23,
and
left pulmonary nodule. C/lest,
21.
J., NOLKE,
BERNSTEIN, Extrapulmonic
vein 1971,
stenosis
A. C., and of
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J. 0.
REED,
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24.
Dilated
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in
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varicos-
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rupture
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Path., 1933, 15, 227-237. KOZUKA, T., and TADAHARU, N. Pulmonary vein anomaly: unusual connection and tortuosity of right lower lobe vein. Brit. 7. Radiol., 1968, LUCAS,
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R. V. Congenital obstruction.
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2975
lungs.
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whom
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rarely,
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van-
failure. Only
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asympto-
free
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in
are
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32.
33.
IKKOS,
J.
YANNACOPOULO5,
Various
D.,
Forms
of Pulmonary
K., and
ALKALAIS,
Pulmonary
varicosi
associated with other congenital abnormalities. Chest, 3972, 62, I07-I09. PERRET, L., and FORTELIUS, P. Ruptured aneurysm of pulmonary vein. Acta tuberc. scandinay., 1961, 4!, 53-55. PERROTT, W. W., and Soo SHIN, M. Pulmonary varix. 7. Thoracic Cardiovasc. Surg., 1974, 68,
35.
S.,
POLLER,
of aorta.
M.
WHOLEY,
A.
stenosis lation, SINGSHINSUK, LIOTT,
veins.
Thorax,
1970,
25,
26, 574-58
41.
and
FRANCIS,
1974,34,
237-239.
A.
S.,
KINKAID, 0. W., and W. H. Selective angiocardiography in diagnosis of varicosity of pulmonary veins: report of case. Am. Heart 7., 1963, 66, 396398.
EL-
M., radiologic
VIAMONTE,
and evaluation
Radio/.
normalities.
J. R. Pitfalls mediastinal abNorth America, 1968,
LEPAGE,
JR.,
of
C/in.
6, 5 1-465. 42.
WAGENvOORT,
J. pulmonary
G. S., MACNEIL, W. V. R. Single left pulvein with normal pulmonary venous roentgenographic curiosity. Am. 7.
VENGSARKAR,
in
97-300.
F.,
D. G.,
VIEWEG,
WEIDMAN,
I.
S., HARTMANN, A. L. P. Stenosis of individual Radiology, 1966, 87, 514-515.
947-952.
J., and
monary drainage: Cardiol.,
Pulmonary
AMPLATZ, K., ANDERSON, R. C., P., and EDWARDS, J. E. Congenital of individual pulmonary veins. Circu-
1962,
JO!,
TRETHEWAY,
D.
J. D.,
ADAMS,
37.
and
varix: evaluation by selective pulmonary angiography. Radiology, 1966, 86, 1078-1081. RIZK, G., MELHEM, R., and DAGHER, I. Bilateral pulmonary varicosities associated with coarcSHONE,
1967,
40.
tation 36.
GENOL.,
39.
889
I. Pulmonary vanices mistaken for and hilar disease. AM. J. ROENTRAD. THERAPY & NUCLEAR MED.,
STEINBERG,
pulmonary
ty
318-320.
34.
38.
Vanices
E.
veins.
C. A., HEATH, Normal development In: The Pathology
Vasculature. Ill., 1964,
Charles
p.
4I0-4II.
D., and EDWARDS, of pulmonary
of the C Thomas,
Pulmonary Springfield,
4
No.
225,
THE
VARIOUS
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REPORT
OF
YORAM ALBERT
By
FORMS
THREE
NEW
BEN-MENACHEM, N. BREST, M.D.,!I
0.
OF
CASES
PULMONARY
AND
REVIEW
M.D., t KOSON KURODA, PRESTON COPELAND,
HOUSTON,
TEXAS
AND
VARICES* OF
M.D.,
E. and
M.D.,t
PHILADELPHIA,
THE
LITERATURE
ROSS
KYGER,
JOHN
III,
D. COAN,
M.D.,
M.D.t
PENNSYLVANIA
ABSTRACT:
True
pulmonary
vanices
are
congenital
local
dilatations
veins, with normal or collateral transpulmonary Pulmonary vanices do not cause pulmonary varices can further become dilated by pulmonary valve disease. Embryologically, venous components an individual unobstructed True
over
pulmonary transpulmonary
pulmonary
the
which collateral
do
usually,
not
only
42
occurred drainage
the in
flew2-4’8-’O’’2’’5-_’7’’9-21’22’23’26’
the
monary
varices.
two
main
REPORT
categories
OF
of
or
hypertension
due
a time into the
primitive system,
that left do
not
enabled atrium. change
to mitral splanchnic atresia
or
adoption in
of of
diameter
treatment.
pattern.
During
dilated,
tortuous
the right the right tered the visualization
and three doubtful2”4 cases were published. Review of the literature revealed differences in the pattern of the pulmonary vanices in several cases, possibly reflecting basic differences in the formation of varices in the embryo. The purpose of this paper is to present three cases and discuss the differences
vein
atrium. but existing
residual venous
symptoms,
require
29-35,38-40
between
at
a pulmonary
into the left hypertension,
venous
may represent the pulmonary
produce
do not
varices are rare. Since of a patient by Hedingen
report
190718
and,
varices into
vein
varices
years,
P ULMONARY first
pulmonary incorporated
of
drainage venous
the
venous
pulmonary
phase
veins
(Fig.
were
2B),
seen
in
lower lobe; these veins drained into superior pulmonary vein, which enleft atrium normally. There was no of a right inferior pulmonary vein.
pul-
CASES
CASE I. H.W., a 50 year old female, was referred for a routine examination because of pulmonary tuberculosis in a member of her family. A chest noentgenogram (Fig. i) revealed dilated vascular
structures
in the
right
lower
lung
field
medially. Review of previous roen tgenograms of the chest showed that these vascular shadows have existed, unchanged, for the past 24 years. The patient was asymptomatic and there was
no evidence ease. evidence
pertension. angiogram *
of congenital
Pulmonary
From
of Radiology,
of
or acquired
angiography pulmonary
The arterial (Fig. 2A) the Departments and
Medicine,
arterial
phase revealed
not
or
venous
disreveal
hy-
FIG.
of the pulmonary a normal atrial
of Radiology,t Thomas
heart
did
i.
Case
vascular field and
i. Chest roentgenogram. markings in the right lower right hilum.
and Surgery,* The University of Texas Medical
Jefferson
University
Hospital,
88i
Philadelphia,
School
Pennsylvania.
at Houston,
and
Prominent medial lung
the
Departments
Ben-Menachem
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882
FIG.
2.
Case
the right
Selective
right
pulmonary
superior
pulmonary
pulmonary
vein.
old female,
II.
R.W.,
a 53 year
clinical
suspicion
space-occupying
lesion.
revealed
aneurysm
carotid (Fig. , lobulated, Pulmonary
a berry
of
an
Cerebral of
right
was
arterial
During the pulmonary
arterial arteries
Case
II.
or
phase were
venous
the
angiography the
left
internal
Note
-
lobulated
into
monary
vein
atrium. CASE
III.
Angiography grams
in the soft
tissue
phase
the
which
phase
drain
E.V.,
a 62
showed 6,
A
(A) posteroanterior mass in the right
were
varicose, side
evaluation
5, A and
lung
drained
left
parathyroid
(Fig.
arterial
varices
(Fig.
right
a single, The
for
The
1975
into
vein.
from
draining
inferior
position. (A) lower pulmonary
the venous
mitted
hypertension.
of the chest, round,
c
veins
left
(Fig. 4, A and B), the found to be normal.
Roentgenograms
anterior
During
ad-
intracranial
artery. Roentgenograms of the chest A and B) revealed a rounded, slightly soft tissue density in the right hilum. angiography showed no evidence of
pulmonary
FIG. 3.
angiogram,
DECEMBER,
(B) During the venous phase, large, There is no right inferior pulmonary
arteries.
for
CASE mitted
i.
normal
shows
et al.
normally
was year
of
common
all
B),
found
to
be
pulinto the
normal.
old
female,
was
ad-
the
left
hyperparathyroidism.
an gland. and B)
and (B) hilum.
adenoma Chest
revealed
lateral
of
roentgeno-
a round,
projections.
VOL.
No.
125,
The
4
Various
Forms
of Pulmonary
883
Varices
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V
4. Case
FIG.
II.
(A) Midstream angiogram,
anteroposterior arterial
and (B)
phase.
Note
slightly lobulated, soft tissue density in the right hilum. Pulmonary angiography showed no evidence of arterial or venous pulmonary hypertension. During the arterial phase (Fig. 7, A and
B)
normal
arterial
architecture
was
found. B), the
During the venous phase (Fig. 8, A and veins of the right lower and middle lobes were found to be draining into a varicose, right inferior pulmonary vein which drained normally into the left atrium. The right superior pulmo-
nary vein normal.
and
both
left
pulmonary
veins
were
varix ment the
anterior oblique selective pulmonary arteries.
normally
the
draining
absence
tension
due
of by
According
to
of
pulmonary
a segenters but
veins;’0
venous
disease,
stability
the over
in
hyperlesions
many
are years
9,12,14,16,17,19,29,32
the
this
7
definition,
cases as well
at
hitherto as our
least
pub-
own
Cases
and II, are not true varices, as they all show anomalies in the involved pulmonary veins, even though the final drainage is into the left atrium.
i
DEFINITION
traditional
pulmonary
pulmonary
to heart
characterized of observation.”
seven
right
is a localized enlargement of of a pulmonary vein which left atrium normally,2 or prominent,
lished,2’4’20’23’26’31’9 DISCUSSION
The
right normal
definition
of a pulmonary
r
r FIG.
.
Case
oblique
ii.
Venous
projections.
phase
Large
pulmonary hilar varices
of
angiogram drain the
in the (A) anteroposterior entire right lung into the left
and atrium
____________________ (B)
right anterior superiorly.
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884
ETIOLOGY
It is generally agreed that pulmonary varices are local congenital anomalies without significant relation to other existing congenital cardiac, pulmonary, or other disorders, and without known etiology.2”30’32’34 The development of the pulmonary venous system and its anomalies has been extensively studied.”6’24’25’28’42 Most probably the defect occurs at the time of transition of the lung from splanchnic venous drainage to pulmonary venous drainage of
et al.
Ben-Menachem
proper,
the
embryonic
ence of tributed these
during
horizons
development.
local varicose to weakness
segments.’
veins
pulmonary as a possible
venous cause
in
the
system of such
is, however, no evidence walls of the splanchnic It is hard basis of
than
The
segments of the venous
Incorporation
splanchnic
weaker
xIIi-xvI28
those
to explain differences
of primitive
newly
developing
was suggested weakness;2 there
to suggest veins are
of the
atin
that the basically
pulmonary
the dilatation in wall structure.
veins. on
the In
cases, in which the of the varicosities
layers,
including
found cases
the
1975
microscopic strucwas studied, all elastic
fibers,
were
be normal.9”8’22’27’29 Only two reported as showing an abnormality: in one,3’ the wall was thinner than normal and the muscular layer was replaced by fibrous tissue; in the other,32 the wall was very thick and fibrous.
to were
DISTRIBUTION
Sex the
existwas walls
most ture
DECEMBER,
and
Age.
literature
three
Among cases reported in females, 19 males, and sex was not reported),2’4”5 (28
whose
the
varices
were
32.2
years
mean
in
years In
mean a small
in the series
females. such as
ences
in age
and
first
sex
discovered
the
one,
are
most as the
age
37.5 differ-
cannot
is usually
an incidental
finding
tigations
for
unrelated
conditions.
Localization. varices were
In found
most in the
and
this
distribution
regarded as significant and ably coincidental, particularly
at
males
during
be probvarix inves-
reported cases, right lower and
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VOL.
No.
225,
middle lobe tional hilar vein lung.
right
not
understood
upon
upper
in this
cases monary THE
did
11.
VARIOUS
True majority
patients),
lobe
and
and
will
paper.
In
have
PATTERNS
DILATED
OF
Forms
of Pulmonary
Varices
885
left upper Ten addias having as to the an entire sparing of
lower
lobe
is
be speculated of the
reported
bilateral
PULMONARY
PULMONARY
Pulmonary of the and
left
not
none
the patient varices.
AND
30
Various
lobe (20 patients) or the and lingula (i. patients). patients were reported varices, without specification involved, or varicosities over The reason for the relative
the
the
The
4
pul-
VARICES VEINS
Varices. These are reported cases (about our
Case
III
belongs
in
this group. The only anomaly is a locally dilated pulmonary vein or veins in part of a lung or its hilum. Two pulmonary veins exist on each side and all enter the left atrium normally. We shall assume that this anomaly may be the result of incorporation of primitive splanchnic venous segments in the newly developing pulmonary venous system, as proposed by Bartram and Strickland.2
B. Dilated Transpulmonary 1”enous Collaterals. Seven patients2’4’”31’39 and our Cases I and ii are recorded as having a single right or left upper or lower pulmonary vein, with dilated, tortuous (varicose) veins draining the rest of the lung into them. The single pulmonary vein enters the left atrium normally. This type of anomaly can theoretically be created by secondary atresia of the pulmonary vein after the separation of the splanchnic venous drainage, but before lobar separation of the pulmonary veins was completed, thereby allowing transpulmonary collaterals to develop. Had the atresia occurred earlier, pulmonary-splanchnic drainage would have been permanently established; a later occurrence would have resulted in individual pulmonary vein stenosis with all its symptomatology and sequelae.5’24’25’36’37 C. Dilated Pulmonaty 1eins with Abnormal Insertion into the Left Atrium. Two
FIG.
7. Case gram, arterial right anterior nary arteries.
Selective
In.
right
pulmonary
angio_ and (B)
(A) Anteroposterior projections. Normal
phase. oblique
pulmo-
cases are reported, one with a left superior pulmonary vein26 and one with a left common pulmonary vein,20 inserting too far to the
right
veins
as true
into
were
the
dilated
varices.
left they
atrium. cannot
Although be
regarded
the
Ben-Menachem
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886
et al.
2975
DECEMBER,
varices. Whether the dilated veins were true varices or not is difficult to say on the basis of the published material; and in the presence ofmitral stenosis, their bilaterality and hilar location suggest that they were probably chronically distended main pulmonary veins rather than true varices. Bryk7 describes seven patients with mitral insufficiency, and with markedly dilated right pulmonary veins. The localization of the dilatation in the right veins in Bryk’s cases can be explained by the direction of the regurgitation flow, which would be preferentially into the right pulmonary veins. These, also, are not true pulmonary varices. SYMPTOMS
When pulmonary varices are the patient’s only abnormality, they do not produce symptoms.2’3”3 In most reported cases the discovery of the varix was on routine chest roentgenograms or during investigation of cardiac or other diseases. Hemoptysis was reported in five patients.2”8’27’3’
In
one”
it
originated
from
bronchiectasis; in another27 bleeding may have been due to pulmonary tuberculosis. Three 1829 presented with cough, which cannot be shown as being directly
8. Case
FIG.
gram,
in
anterior
pulmonary
In.
the
Venous phase of pulmonary angioanteroposterior and (B) right
(A)
oblique vein
into
the left atrium.
vein
is normal.
projections. is varicose,
The
The right but drains
right
superior
inferior normally
caused
by
the
with
congestive
causes,
pulmonary
THE
but
heart unrelated
RELATIONSHIP
“Pulmonary
Varices”
described mitral
a patient stenosis and
Viamonte
in Mitral and
StenoLePage4’
(their Case II) with bilateral pulmonary
of
OR
Most alies disease:
commonly, were
pul-
ciency;8”9’20’21’33
patients three
various
varices. ACCOMPANYING
DISEASES
AND
VARICES
accompanying
congenital five
of
the
BETWEEN
PULMONARY
D.
failure to
ANOMALIES
sis andlnsufficiency.
presence
monary varices. One patient23 presented with a middle lobe syndrome, directly related to pressure on the middle lobe bronchus by the varices. Two patients presented with dysphagia,20’26 due to pressure on the esophagus by a large left pulmonary vein which inserted abnormally to the right of the left atrium. Both these patients are not regarded as having true varices. Most symptomatic patients presented
anom-
or
acquired
heart
had
mitral
insuffi-
had
mitral
steno-
VOL.
225,
No.
The
4
Various
Forms
of Pulmonary
and one each had coarctation of the aorta,’5 aortic stenosis and pseudocoarctation,2 common origin of both great vessels from the right ventricle,’8 ventricular septal defect and patent ductus arteriosus,4#{176} malrotation of the cardiac chambers,2 and absence of the right hemipericardium.3#{176} Other associations included pulmonary tuberculosis,27 systemic hypertension,22 left upper lobe pulmonary emphysema,2 intracranial aneurysm (our Case ii), and hyperparathyroidism (our Case III). Three patients had pulmonary arterial anomalies, including absence of the right main pulmonary artery,’2 hypoplasia of the descending branch of the right pulmonary artery,” and a trifurcation, rather than bifurcation, of the right pulmonary artery.3 Two patients2 had partial anomabus venous drainage contralateral to the side of the pulmonary varices. Varices are not created by heart disease, and their existence in cardiac patients is purely coincidental.2” In patients with pulmonary venous hypertension due to mitral stenosis or insufficiency, pre-existing varices may become wider.” In such cases, progression of the congestive heart failure may make the presence of an enlarging varix rather threatening,’ by creating conditions for rupture. In one patient with mitral insufficiency’9 the varices became progressively distended over a seven year period and disappeared following surgery. In another,2#{176} dysphagia due to a distended left pulmonary vein with an aberrant insertion into the left atrium and pressure on the esophagus disappeared after treatment of the patient’s congestive heart failure.
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sis;8.9.16
COMPLICATIONS
Two patients29”2 suffered a cerebral which was possibly Hedinger’8 felt odenal ulcer was from
deaths from
DEATH
are reported to have embolus, the source of in the varices. that his patient’s ducaused by an embolus
a pulmonary
Three sulted
AND
reported ruptured
as having pulmonary
re-
887
varix.’”2 In one, the varix grew rapidly with aggravation of existing right heart failure. In another, rupture may have been caused by erosion of the varix by pulmonary tuberculosis in the adjacent lung tissue. The complete clinical picture of the third patient’2 is not available. A fourth patient29 is reported to have died of a cerebral embolus, but the description of the clinical picture in this patient is strongly suggestive of congestive heart failure. ROENTGENOGRAPHIC
DIAGNOSIS
DIFFERENTIAL
AND
DIAGNOSIS
The smooth, rounded or lobulated soft tissue density of the varix on the plain chest roentgenogram and tomogram should be differentiated from other pulmonary or hilar mass lesions, including those that are neoplastic, granulomatous, lymphatic, or vascular in origin.2” 8,10, 1316,19.20,21,38 At least one patient’4 was given treatment for two years for pulmonary tuberculosis, until the true nature of the mass on the chest roentgenogram was revealed. A most important differential diagnosis is that of pulmonary arteriovenous 1330 which may have an identical roentgenographic appearance. Partial anomalous pulmonary venous return should also be included in the differential diagnosis.’9 The Muller maneuver enlarges the size of pulmonary varices, and the Valsalva maneuver decreases it in many, but not all patients.2”6”9 The only safe criterion for the diagnosis of pulmonary varices is the opacification of the varices during the venous phase of pulmonary angiography. Demonstration
of
normal
pulmonary
ar-
terial and venous pressures is necessary to exclude heart disease as an etiologic factor. An important additional criterion is the non-growth
of
pulmonary
varices
over
long periods of follow-up. Eleven tients3’9”2”4”6”7”9’29”2 and our Case follow-up chest roentgenograms over ods
varix.
are a
Varices
ranging
from
two
any appreciable change pulmonary varices.
to
24 years,
in
the
I
pahad peri-
without
size
of
the
et al.
Ben-Menachem
888
the
TREATMENT
Treatment
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matic
is not
patients
disease.’4’21”#{176}’3’ failure, ces, the
necessary
who
who
with
heart
of the
reduce of
14.
heart
right
enlargement
can be expected to lesions with treatment
tive
of
the size of their conges-
15.
hemoptysis
as
in
exists
pulmonary
the
and
patient
in
is directly
varices,
related
should
be 17.
considered.2 Yoram Department
of
Hermann
Ross
3203
Radiology
Houston,
I 8.
Sterling
AUER,
77025
19.
2.
J.
of
variations.
Jo!, BARTRAM,
D.,
7. Canad. 4.
human
20.
pulmonary
Anat.
Rec.,
BENFIELD,
A.
Radiologists,
J. R.,
GoTs,
Anomalous single ing parenchymal
1972,
R. E.,
23,
and
left pulmonary nodule. C/lest,
21.
J., NOLKE,
BERNSTEIN, Extrapulmonic
vein 1971,
stenosis
A. C., and of
23.
J. 0.
REED,
pulmonary
veins.
24.
Dilated
J. E.,
HASTINGS, nary
varix:
49,
1974,
II.
Editorial. 1966,86,
12.
ELLIS,
veins
in
26.
varicos-
1951,
F. M.,
KELVIN,
245,
25,
FRASER,
pulmonary
with
PAR,
veins.
In:
and
Angiology,
J. A. P. Varicosities Diagnosis
of Disease
and HUNT, spontaneous
A.
D.
Radiologists,
H.
D.
Pulmonary
rupture
of
death.
Path., 1933, 15, 227-237. KOZUKA, T., and TADAHARU, N. Pulmonary vein anomaly: unusual connection and tortuosity of right lower lobe vein. Brit. 7. Radiol., 1968, LUCAS,
4!,
232-234.
R. V. Congenital obstruction.
causes Cardiovasc.
of pulmonary C/in., 1972,
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J. H., KANJUH, V. I., and E. Anomalies of pulmonary veins. Am. 7. Cardiol., I967, 20, 77-90. NASH, B., FREIDENBERG, R. M., and GOETZ, R. A.,
NAKIB,
MOLLER,
J.
insertion
of pulmonary
simulating
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AM.
NAUwERK,
intrinsic
vein
lesion
ROENTGENOL.,
RAD.
MED., 1961, 86, D. Lungenvarix Med. Wochenschr.,
888-891.
NEILL,
und
into
left
of esopha&
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B. H. Varix
&
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AM.
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32,
608-612. 30.
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W.
of
Varicosities arteriovenous
of
13-17.
428-431.
R. G., and
7. Canad.
225-228.
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Pediatrics,
CLEVELAND,
lung
pulmo-
45,
3972,
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CHAIT,
distended
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W., and
K., and
227-229.
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BOONE,
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23,
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1011-1012.
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JACOBS,
EDWARDS,
24-27.
GOLDEN, M. S., PRICE, H. L., J. E., and CHEITLIN, M. D. Pulmodiagnostic pitfall. Circulation,
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