CASE
REPORTS
One-Stage Surgical Treatment
of
Cardiac
and
Pulmonary
Echinococcosis
Alejandro
Aris,
M.D., Carlos Leon, M.D.,
Jose
0.
Bonnin,
M.D.,
Constantino
Serra,
M.D.,
and
Jose
M. Caralps, M.D.
ABSTRACT
The case of
a
26-year-old woman
with
cardiac and pulmonary echinococcosis is presented.
Surgical treatment of the intrathoracic disease was
done in a one-stage operation. Under cardiopulmo-
nary bypass, a left ventricular hydatid
cyst
and
a
lower lobe pulmonary cyst were removed. The pa-
tient is completely asymptomatic and is leading
a
normal life two years after operation.
Cardiac echinococcosis represents an infre-
quent complication of hydatid disease but its
effects can be devastating if not treated aggres-
sively. The purpose of this article is to report
the case of a young woman with previously
treated hepatic echinococcosis in whom pul-
monary and cardiac hydatid cysts developed.
The patient underwent successful surgical re-
moval of the cysts in a one-stage operation with
cardiopulmonary bypass.
A
26-year-old woman was admitted to another
hospital because of shortness of breath and car-
diac palpitations following mild exercise. The
episode was diagnosed as supraventricular
tachycardia and was treated by electric counter-
shock, which was successful. Pertinent past
history included two abdominal operations be-
cause of hepatic hydatid cysts,
15
and
7
months
previously.
A
routine chest roentgenogram was
found to be abnormal. The diagnosis of cardiac
and pulmonary echinococcosis was suggested,
and the patient was transferred to our hospital
for further workup and therapy. Chest roent-
genogram made at admission (Fig
1)
revealed a
mass at the left heart border as well as a solitary
bilocular nodule at the posterior aspect of the
left lower lobe. The electrocardiogram showed
From the Cardiac and Thoracic Surgery Units, Hospital de
la Santa Cruz
y
San Pablo, Barcelona, Spain.
Accepted for publication June
10, 1980.
Address reprint requests to Dr. Aris, Chief, Cardiac
Sur-
gery Unit, Hospital de la Santa Cruz
y
San Pablo, Avda
S.
Antonio
M.
Claret
167,
Barcelona 25, Spain.
Q
waves and negative
T
waves in leads
I
and
aVL, suggestive of myocardial necrosis of the
high lateral wall (Fig 2).
Laboratory tests were essentially normal ex-
cept for mild eosinophilia and a positive sero-
logical test for hydatid cyst (latex agglutina-
tion test). The echocardiogram indicated the
presence of a mass near the base of the aorta.
The patient underwent cardiac catheteriza-
tion. Left ventricular end-diastolic pressure was
18
mm Hg. Left ventriculogram showed hy-
pokinesia of the lateral wall. Coronary arte-
riogram revealed no obstructing lesions but
the two branches of the left coronary artery
were displaced by a mass, which created a
"cold zone" between them (Fig
3).
The diagnosis of left ventricular hydatid cyst
and left lower lobe hydatid cysts was made, and
the patient underwent operation. The chest was
entered through a transsternal bilateral thora-
cotomy, which provided ample exposure of the
heart and both pleural cavities. While the aorta
and venae cavae were being cannulated for car-
diopulmonary bypass, a rapid supraventricular
tachycardia developed. Once the patient was on
the pump, the pericardium was freed from the
lateral, upper aspect of the left ventricle where
an egg-sized mass, covered by some myocardial
fibers, was bulging.
The cyst was punctured, and its contents
were aspirated with a syringe.
A
creamy yellow
material was obtained. Then the operative field
was covered with gauze pads moistened with
3%
saline solution. The cyst was opened, and
a large number of ruptured membranes of
daughter cysts were removed. The cavity was
emptied, and most of the fibrous pericyst was
excised. The deepest part was left untouched
since there was only a thin layer of myocardium
between it and the left ventricular cavity. The
heart was defibrillated, and while the patient
was still on the pump, the entire left lung was
retracted medially.
A
bilocular mass was iden-
564
OOO3-4975/81/060564-05$01.25
@
1980
by The Society
of
Thoracic Surgeons
565
Case
Report:
Aris
et
al: Cardiac
and
Pulmonary Echinococcosis
A
Fig
1.
(A)
Posteroanterior chest roentgenogram shows a
mass at the left heart border and
a
nodule at the left
lung field.
(B)
Lateral view shows this nodule located
posteriorly
(arrow).
Fig
2.
Electrocardiogram shows
Q
waves and negative
T
waves in leads
1
and aVL, which suggest necrosis
of
the
high lateral wall.
B
tified in the lower lobe and excised en bloc.
Following the cystectomy, capitonnage of the
residual cavity with interrupted sutures of ab-
sorbable material was performed. Cardiopul-
monary bypass was discontinued and after
careful hemostasis, the chest was closed. The
patient made an uneventful recovery and was
discharged on the tenth postoperative day.
Follow-up at regular intervals reveals that she
has not had any further arrhythmias and
is
leading a normal, active life.
Pathological examination of both specimens
I
II
Ill
aVR aV
L
aVF
v1 v2
v3
v4
VS
V6
566
The
Annals
of
Thoracic
Surgery
Vol
31
No
6
June
1981
Fig
3.
Left coronay arteriogram in left anterior oblique
projection. The left anterior descending corona
y
artey
is displaced upward and the circumflex corona
y
arte
y,
posteriorly. There is an avascular zone in between
them.
was consistent with hydatid cyst. Necrotic ma-
terial was found in both cavities but no scolices
were seen.
Comment
Different reports [l-31 estimate that 0.5 to 2% of
all cases of hydatid cysts involve the heart. Most
of the cases occur in countries where the dis-
ease
is
endemic-Uruguay
[l,
4-81, Argentina
191, Iran
[lo,
111,
Greece [12,131, Israel [14,151,
and Spain. In the last named, more than 20
cases have been reported, most of them by sur-
gical teams [16-201.
Implantation of the hexacanth embryo in the
myocardium usually occurs after the embryo
has passed the pulmonary capillary network
and has reached the heart by way of the coro-
nary circulation. This explains the higher inci-
dence
of
echinococcal cysts in the left ventricle
and ventricular septum, which have the richest
blood supply. Before it reaches the heart, the
embryo of the
Echinococcus
grunulosus
has fil-
tered through the sinusoids of the liver (the
most commonly involved organ) and the pul-
monary circulation. The embryo may be im-
planted in these organs, developing an hydatid
(or echinoccocal) cyst. This sequence of events
occurred in our patient, in whom hepatic, pul-
monary, and cardiac hydatid cysts developed in
the short interval of 15 months.
The fate of a cardiac echinococcal cyst is usu-
ally rupture, either in the pericardial cavity [21,
221 or in a cardiac chamber (38% of the cases
according to Di Bello and Menendez [7]). When
the latter occurs, death of the patient by
anaphylactic shock or massive embolization to
different organs
[ll,
22-24] can ensue.
Cardiac hydatid cysts rupture more fre-
quently than do hydatid cysts in other organs,
probably because of the constant motion.
Also,
rupture in the right ventricle is more frequent
than in the left (88% versus 33%) [71 because of
the thicker wall and higher pressure in the lat-
ter. Other events in the life of a cyst include
rupture inside the adventitia with formation of
daughter cysts, and interference with the con-
duction system of the heart, thereby inducing
arrhythmias [25]. Both were found in our pa-
tient.
Diagnosis of cardiac hydatid disease is dif-
ficult, especially in the early stages. Roent-
genographic findings can be consistent with
an aneurysm 15, 261. Although several changes
in the electrocardiogram are said to be charac-
teristic of cardiac echinococcosis [2, 8, 141 and
helpful in distinguishing it from ventricular
aneurysm, our patient showed
Q
waves in
some leads, indicating myocardial necrosis,
an unusual feature in cardiac hydatid disease.
Echocardiography has proved a valuable diag-
nostic test in detecting the location of possible
cardiac cysts [18], as it did in our patient.
Cardiac catheterization with cineangiog-
raphy is the most reliable test for an accurate
diagnosis. Coronary angiography also should
be performed since coronary compression by a
growing cyst has been reported
[17].
In our pa-
tient, the lumen of the arteries was patent but
the cyst had displaced the two branches of the
left coronary artery, thereby creating an avas-
cular zone between them. This angiographic
finding was described in 1976 [271 and 1979
1201.
Cardiac echinococcosis should be treated
surgically as soon as the diagnosis is estab-
lished because of the fatal complications that
can occur. Intervention involves the removal of
567
Case Report:
Aris
et
al:
Cardiac and Pulmonary Echinococcosis
the cyst (or cysts), including the adventitia.
Needle aspiration prior to the surgical excision
is advisable and if clear liquid is obtained, the
cyst should be injected with a sterilizing solu-
tion (hypertonic saline, ether, formaldehyde)
before it is opened.
Although most of the reported patients with
cardiac echinococcosis were operated on
through a.left thoracotomy without the aid of
cardiopulmonary bypass [3,
10,
12-15], the
low-risk perfusion technology now available
makes the use of extracorporeal circulation ad-
visable.
Regarding pulmonary hydatid cysts, the
history of our patient is fairly representative.
The disease usually is located in one lobe. In a
series of
100
patients reported by Aytac [28], the
distribution of single-lobe involvement was
about the same for the left upper and both
lower lobes. Symptom-free patients are rare
(about
5%)
[28, 291, and pulmonary involve-
ment in our patient prabably would have
passed undetected if the cardiac cyst had not
developed. Enucleation of the cyst [30] followed
by capitonnage of the cavity is the preferred
treatment for small, noncomplicated
Echinococ-
cus
cysts of the lung [28].
The case of our patient deserves some par-
ticular comments. The first symptoms of the
disease were arrhythmias, a rare presentation.
According to Heyat and associates 1101, who re-
viewed the signs and symptoms in 82 patients
with cardiac hydatid cysts, only
11%
had “pal-
pitation of the heart.”
The existence of pulmonary and cardiac
echinococcosis in the same patient has been re-
ported previously
[ll,
181 but, we believe that
we are among the first to remove both cysts in
a
one-stage operation. A second operation to re-
move the lung cyst was avoided.
Although there is a recent trend to use me-
dian sternotomy for pulmonary operation, this
approach has been questioned for lesions in-
volving the left lower lobe [31]. For this reason,
we decided to use a transverse transsternal bilat-
eral thoracotomy, which provided excellent ex-
posure of the heart as well as of
the
posterior
aspect of the left lower lobe. Under cardiopul-
monary bypass, the entire left lung was col-
lapsed and the pulmonary cyst easily excised.
Our patient has recovered completely and is
free from hepatic, pulmonary, and cardiac
echinococcosis two years following the in-
trathoracic procedure.
References
1.
2.
3.
4.
5.
6.
7.
8.
9.
10.
11.
12.
13.
14.
15.
16.
Dighiero
J,
Canabal
EJ,
Aguirre CV, et al:
Echinococcus disease
of
the heart. Circulation
17:127, 1958
Murphy
TE,
Kean BH, Venturini A, Lillehei CW:
Echinococcus cyst of the left ventricle: report
of
a
case with review of the pertinent literature.
J
Thorac Cardiovasc Surg
61:443, 1971
Ramos G, del Villar
JL,
Sainz
JL,
et al:
Hidatidosis cardiaca. Rev Clin Esp
121:411,1971
Artucio H, Roglia
JL,
Di Bello
R,
et al: Hydatid
cyst
of
the interventricular septum of the heart
ruptured into the right ventricle.
J
Thorac Car-
diovasc Surg
44:110, 1962
Di Bello
R,
Rubio
R,
Dighiero
J,
et al: Pseudo-
aneurysmatic
form
of cardiac echinococcosis: re-
port
of
a new case and review of the literature.
J
Thorac Cardiovasc Surg
45:657, 1963
Di Bello
R,
Ab6 JC, Dubra
J,
Diaz EG: Hydatid
cyst of the heart.
J
Thorac Cardiovasc
Surg
46:522, 1963
Di Bello
R,
Menendez H: Intracardiac rupture of
hydatid cysts of the heart: a study based on three
personal observations and
101
cases
of
the world
literature. Circulation
27:366, 1963
Canabal
EJ,
Dighiero
J,
Purcallas
J,
et al:
Echinococcus disease of the left ventricle: a clini-
cal, radiological and electrocardiographic study.
Circulation
12:520, 1955
Gonzalez Bosch
R,
Mosto D: Hidatidosis car-
diaca. Prensa Med Argent
24:38, 1937
Heyat
J,
Mokhtari H, Hajaliloo
J,
Shakibi JG:
Surgical treatment
of
echinococcal cyst of the
heart.
J
Thorac Cardiovasc Surg
61:755, 1971
Handjani AM, Farpour A, Mechanic
K,
et al:
Cardiovascular echinococcosis. Am
J
Surg
117:666, 1969
Karageorgis
B,
Papanicolis
I:
Some remarks on
two
personal cases
of
cardiac echinococcosis. Dis
Chest
51:199, 1967
Papamichael
E,
Ikkos
D,
Milingos M, Yan-
nacopoulos
J:
Echinococcosis of the heart. Chest
59:280, 1971
Romanoff H, Milwidsky H: Primary echinococ-
cosis of the heart cured by operation.
J
Thorac
Cardiovasc Surg
43:677, 1962
Romanoff H: Echinococcosis
of
the heart: report
of three new cases.
J
Thorac Cardiovasc Surg
66:29, 1973
Calvo Melendro
J:
Quistes hidatidicos de cora-
z6n. Rev Clin
ESP
42:397, 1951
17.
Rivera
R,
Delcan JL: Surgical treatment of coro-
568
The
Annals
of
Thoracic Surgery
Vol
31
No
6
June
1981
18.
19.
20.
21.
22.
23.
24.
nary insufficiency produced by cardiac echino-
coccosis. Chest
78:849, 1980
Soler-Soler
J,
Noguera
L,
Fit0
R:
Recent multiple
pulmonary nodules with progressive cardio-
megaly. Chest
73:397, 1978
Tellez G, Nojek C, Juffe A, et al: Cardiac
echinococcosis: report of
3
cases and review of
the literature. Ann Thorac Surg
21:425, 1976
Montoyo JV, Nogues-Antich FJ, Rivas
L,
et al:
Left ventricular echinococcosis diagnosed by
coronary cineangiography. Eur
J
Cardiol
10:215,
1979
Di Bello
R,
Stanham
J,
Rubio
R,
Muxi F: Hydatid
cyst of the left ventricular wall with rupture into
the intrapericardial space.
J
Thorac Cardiovasc
Surg
44:268, 1962
Purriel P, Perez Fontana
V,
Muras
0,
Mendoza
D: Equinococosis cardiaca con siembra pericar-
dica y de pequeiio y gran circulo. Torax
2:188,
1953
Dumolard MM, Lemaire C: Kyste hydatidique de
l’oreillette droite. Alger Med
32:293, 1928
Purriel P, Muras
0,
Tomaluro D, Mendoza D:
Equinococosis cardiaca: siembra metastatica
pulmonar. Torax
1:223, 1952
25.
Peters JH, Dexter
L,
Weiss
S:
Clinical and
theoretical considerations of involvement of the
heart with Echinococcus cysts. Am Heart
J
29:143, 1945
26.
Hatzeganu
I,
Vasiliu
T,
Moga A: Le kyste
hidatidique du coeur simulant un anevrisme.
Arch Ma1 Coeur
29:72, 1936
27.
Fawzy ME: Hydatid disease
of
the heart. Br
Heart
J
38:307, 1976
28.
Aytac A, Yurdakul
Y,
Ikizler C, et
al:
Pulmonary
hydatid disease: report of
100
patients. Ann
Thorac Surg
23:145, 1977
29.
Sarsam A: Surgery of pulmonary hydatid cysts:
review of
155
cases.
J
Thorac Cardiovasc Surg
62:663, 1971
30.
Barret NR: Removal of simple univesicular pul-
monary hydatid cyst. Lancet
2:234, 1949
31.
Urschel HC Jr: Discussion of Cooper JD, Nelems
JM, Pearson FG: Extended indications for me-
dian sternotomy in patients requiring pulmo-
nary resection. Ann Thorac Surg
26:413, 1978
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