A Gyant Splenic Hydatid Cyst Case Presen

m e d i c r i t r e v i s t a de m e d i c i n a i n t e r n a y c r í t i c a
case report
A Gyant Splenic Hydatid Cyst Case Presented with Dyspnea and Fever
Miguel Aquise Chávez, M.D., Ernesto García Vicente, M.D.,
Adriana Aquise Pino, M.D., Mª Raquel Pellicer Algora, D.U.E.,
José Luis Pellicer Espligares, M.D., PhD.
ABSTRACT—This case is based on the definitive diagnosis of a large splenic
hydatid cyst in a patient who presented an unspecific clinical picture though
showed suspicious findings in the first chest radiography performed. The CT
scan images clarified the diagnosis. In the beginning these images induced confusion among experimented clinicians due to their great similarity with pathologic
abdominal processes like the retention stomach or the empty viscera covered
perforation. Subsequently, the diagnosis of a large infected hydatidic splenic
cyst was confirmed. This issue is considered common in our environment but
very unfrequent when the affected organ is spleen.
KEY WORDS—Hydatid disease, spleen, splenic hydatic cyst, splenic hydatidosis,
splenic echinococcosis.
MEDICRIT 2009; 6(1):54-57
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A
59 year - old female , with unremarkable medical and sur -
gical histories except an amigdalectomy in her childhood. She didn´t take
any prescription or over-the counter medications. 6 days before the admission presented a left-sided pleuritic-like pain with subsequent radiation to the
left hemi-abdomen, presenting fever 48 hours later, and developing dyspnea with
minimum effort 72 hours after the fever onset, being this the main reason for
presenting to the Emergency Department.
On physical examination, the patient was conscious, oriented and receptive,
she was of moderate build and appeared healthy and well hydrated, with a plumcolored malar flush. She was in sinus tachycardia, with a heart rate of 110 bpm,
with no murmurs or added sounds, and her blood pressure was noted to be
140/80 mmHg. In the pulmonary auscultation, the vesicular murmur was diminished in 1/3 of the left lower hemithorax. A doubtful abdominal mass occupying
left flank and left hypochondrium exceeding sagittal medial line and leaving the
Dr. Miguel Aquise Chávez. Gastroenterology Department; — Dr. Ernesto García Vicente. Intensive Care Unit; — Mª Raquel
Pellicer Algora. Nursing Department; — Dr. José Luis Pellicer Espligares. Chief of Surgery Department. Santa Bárbara
Hospital. Soria. Spain. — Adriana Aquise Pino. Complutense University School of Medicine. Madrid. Spain. Corresponding
author: Ernesto García Vicente. Avenida de la Constitución nº 10, 1ºB. 42004. Soria. Spain. Phone: +34 607662049. E-mail:
ernesdino@yahoo.es.
This manuscript´s authors declare not to have conflict of interests as well as not having published or submitted any related
papers from the same study. In the same way, this manuscript is not under consideration elsewhere, and we certified that all
authors had access to the data and a role in writing the manuscript.
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m e d i c r i t r e v i s t a de m e d i c i n a i n t e r n a y c r í t i c a
left iliac fossa was palped; rest of the abdomen
without other findings; neither hernias nor adenopathies.
The initial work-up included laboratory investigations without any clinically significant results
except for the urine analysis which developed the
presence of 20-30 leukocytes/field. Basal arterial
gasometry: pH: 7.47; PO2: 56 mmHg; PCO2: 29.4
mmHg; HCO3: 21.3 meq/l; BE:-1; SaO2: 91.6%. A
chest x-ray was performed showing an elevation
of the left hemidiaphragm with left costophrenic
sinus obliteration, and an air-fluid level in the
gastric chamber was observed (Figure 1). The
abdominal x-ray showed a calcium-density image
insinuating the iliopectineal ligament and dilated
colonic haustra (Figure 2).
She was admitted to the internal medicine ward
with the diagnosis of respiratory insufficiency and
urinary tract infection, and a computer tomography scan was ordered, showing anelevation of both
hemidiaphragms with a minimal pleural effusion
(Figures 3 and 4) accompanied by a small atelectasis. In the abdominal study a large mass was
easily observed, with maximum diameters of 17
cms (cephalocaudal), 16 cms (anterior-posterior)
and 12.1 cms (transverse). This mass presented
well formed partially calcified walls and contacted
with the left hemidiaphragm, to which it displaced
and compressed. An air-fluid level with plenty
of gas bubbles and heterogeneous content with
predominance of low density areas could be visualized inside. Hence, everything indicated the
presence of an infected cyst, probably placed in
the spleen, with the morphological appearance
of a hydatidic cyst. With these findings, the patient was admitted to the Department of Surgery.
Two days later new laboratory resuls were
received, emphasizing a high IgE (1190 U/ml)
and PCR levels (356.7 mg/dl) and broad-spectrum
antibiotic prophylaxis with Imipenem (1g/6hs)
was implemented. Three days later the cyst was
punctured and aspired under CT scan control
with a thick needle obtaining a foul-smelling
fluid and in the same procedure a 12 F pig-tail
drain was placed, evacuating between 50 and
500cc/day. Culture of the discharging material
grew a Streptococcus sp. and Echinococcus granulosus.
In a control CT scan performed 4 days later the cyst had maximum diameters of 15 cms
(cephalocaudal), 14 cms (anterior-posterior) and
10.5 cms (transverse).
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1
2
3
4
Figuras 1 a la 4.
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55
a gyant splenic hydatid cyst case
3 days later a surgical programmed intervention was performed (10th day from admission).
A left subcostal laparotomy revealed a 15 x 10
cms infected hydatidic cyst located in the lower
pole of the spleen, containing foul-smelling pus,
hydatidic vesicles and gas. This cyst presented
numerous soft adhesions to epiplon, stomach
and transverse colon. These adhesions were easily
liberated and no splenectomy was needed, and
a 2/3 cystpericystectomy was performed and a
drainage was placed into the area. No postoperatory incidents were reported and the patient was
discharged with no further problems.
discussion
Hydatid disease caused by the genus Echinococcus is endemic in Eastern Europe, Middle East,
South America, North Africa, Indian subcontinent,
Australia and New Zealand.1 Echinococcus granulosus
is the commonest organism involved, with dogs
usually as the definitive host and human beings
acting as an accidental intermediate host.
Enzymatic digestion of the shells of the eggs
(shed in the faeces of the dogs) in the duodenum
of the intermediate host release the embryonic
forms. These migrate through the intestinal wall
to enter the portal circulation, from where they
make their way towards the liver or lung, developing a triple cystic covering which grows grows
at a rate of about 1 centimeter a year.2
Although hydatid disease is one of the oldest
diseases known to man, the incidence of exclusive
splenic location is very low, constituting 0.5 to
4% of all cases of hydatidosis,3 whereas the liver
(70%) and the lung (15%), are the commonest
settlements. The rare sites include heart,4 thyroid,5
breast,6 central nervous system,7 etc. Hydatid cyst
is the only parasitic cyst of the spleen and it is
said to be twice as common as the non-parasitic
variety. Dieulafoy (1898) classification divides
splenic hydatid cysts into ascending (immobile)
and discending (mobile) types and into cortical,
central and juxtasplenic types as per location of
the cyst, being the abdominothoracic type added
by Scherb (1904). Segond and Potherat divided
hydatids into three groups: (a) those containing
much fluid but few daughter cysts, (b) those packed with vesicles and (c) suppurating cysts. The
largest cyst ever reported was in Australia and it
contained 57 litres of fluid.8
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A hydatid cyst consists of three layers. The
outermost layer is the adventitia (pseudocyst),
consisting of fibrous tissue as a result of the
granulomatous reaction of the liver to the cyst,
from which it is inseparable. The middle layer
is the laminated membrane (ectocyst), which is
formed of the parasite itself, and contains a crystal
clear fluid if it is not superinfected. The hydatid
fluid is antigenic and highly toxic, and can cause a
potentially fatal anaphylactic reaction in humans.
The innermost layer is the germinal epithelium
(endocyst), being the only living part of the hydatid
cyst and secreting the hydatid fluid internally and
the laminated membrane externally, producing
the new generations of parasites.
Although various hypothesis about the possible
routes followed by the parasite in order to reach
the spleen have been formuled, including vascular
and linfatic dissemination, reflux into spleen from
portal vein, through haemorrhoidal veins, through
small veins passing from the splenic flexure of
the colon to the lower pole of the spleen, via
arterial blood etc, the most logic route seems to
be escaping of 10-15% of embryos from liver and
lung filters into general circulation, a fraction of
which settle down in the spleen.
The first clinical indication of the presence of
splenic hydatid disease usually is an accidentally
discovered mass in the abdomen mostly in left
hypochondrium and less frequently in the epigastrium, pain which is usually a dull dragging
ache, dyspepsia, constipation due to pressure
on colon, hypertension due to left renal artery
compression, and dyspnoea due to pushing up
of the left diaphragm,9 however up to 30% are
incidental findings in asymptomatic individuals.10
Other forms of clinical presentation include the
rupture of splenic echinococcal cyst with anaphylactic shock11 traumatic or not, acute abdomen,12 rupture of longstanding splenic hydatid
cyst into bronchial tree,13 or even a splenic hydatid
cyst established fistula to the colon.14
Laboratory evaluation of patients with hydatid
disease often yields non-specific data, being eosinophilia (above 3%) not significant in endemic
areas. Elevated levels of IgE levels are a non-specific
indicator of prior sensitization or active infection
with parasitic organisms, while elevated levels of
IgM classes specific to echinococcal organisms
may be a sensitive indicator or recurrent disease.15
Serum immunoelectrophoresis is currently the
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most reliable, with a sensitivity of approximately
90%, with 1 year positivity after the organism
has been eradicated. The Casoni intradermal skin
test, indirect hemagglutinition and ELISA had
also been used.
Image techniques have pushed biochemical
into the background in the diagnosis of this disorder, and despite the fact that the diagnosis
of splenic hydatidosis is difficult or impossible
by conventional radiography alone, if the cyst is
calcified hydatidosis may be suggested. Plain radiology of abdomen can reveal a soft tissue shadow
with or without calcification, displacement of left
diaphragm upwards, stomach to the right and
transverse colon with splenic flexure downwards.
Ultrasound and computed tomography (alone or in
combination) established the definitive diagnosis
of splenic hydatidosis in almost all the cases,
being the echography cost effective and particularly valuable for follow up screening. CT is
more accurate than ultrasound in localizing and
delineating extent of the cyst.
Regarding to the medical treatment, Mebendazole (60 mg/kg/day for 6-24 months) or Albendazole (10 mg/kg/day for 6 months) can only
complement surgery but cannot replace it, though
in the last twenty years the need of systematic
splenectomy in this kind of interventions has been
critically reduced, avoiding overwhelming post
splenectomy infection. Besides, though not in
this case due to the apical location of the cyst,
the laparoscopic surgery has an important and
current role in this issue.16,17
The didactic interest of this case is based on
the final diagnosis of a large splenic hydatid cyst
in a patient who was admitted to the Emergency
Department presenting unusual and unspecific
clinical signs but showing suspicious findings in
the first chest radiography performed being confirmed by spectacular abdominal CT scan images.
In the beginning these images induced confusion
among experimented clinicians due to their great
similarity with pathologic abdominal processes
like the retention stomach or the empty viscera
covered perforation. Subsequently, the diagnosis
of a large infected hydatidic splenic cyst was confirmed, being this issue considered common in
our environment but very unfrequent when the
affected organ is spleen.
It is important to underline that it is relevant
to maintain hydatid disease and its possible complications as one of the differential diagnosis to
consider in endemic areas when the clinical condition justify it, being compulsory the prevention
against this parasitation by Public Health.
references
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