ECHINOCOCCOSIS

Human echinococcosis is a parasitic disease caused by tapeworms of the genus

Echinococcus.The two most important forms in humans are cystic echinococcosis
(hydatidosis) and alveolar echinococcosis.Humans are infected through ingestion of
parasite eggs in contaminated food, water or soil, or after direct contact with animal hosts.
Echinococcosis is often expensive and complicated to treat and may require extensive
surgery and/or prolonged drug therapy.Prevention programmes focus on deworming of
dogs, which are the definitive hosts. In the case of cystic echinococcosis preventive
measures also include, deworming dogs, slaughterhouse hygiene, and public education.
More than 1 million people are affected with echinococcosis at any one time.
Echinococcus is an infection caused by a parasitic tapeworm from
the Echinococcus genus. A few different types of tapeworms can cause echinococcus in
humans, including: E. granulosus, E. multilocularis, and E. vogeli. In some cases, the
organs affected depend on which type of tapeworm has caused your infection.The
infection is rare in the United States. It occurs more often in the Mediterranean, Middle
East, Africa, and Central Asia. If left untreated, it can be fatal. With treatment, your
outlook may be good.
What are the symptoms of echinococcus?

Your symptoms will vary depending on which organs are affected. According to Stanford
University:

The infection affects the liver in about 75 percent of people who contract it. Symptoms may
include pain in your abdomen and the formation of cysts on your liver.

The infection affects the lungs in about 22 percent of people who contract it. Respiratory
symptoms may include chest pain and coughing up bloody mucus.

Other areas of your body can also be affected, including your skin, spleen, or kidneys.

What causes echinococcus?

If a parasitic tapeworm infects you, echinococcus will develop. The parasite enters a host,
which is usually an animal, such as a dog, sheep, or goat. The worm lives in the bowels of the
animal and releases its eggs into the animal’s feces.

You’re most likely to contract the infection when you eat food that has been contaminated
with animal feces. After eating contaminated food, the incubation period is usually a few
months long.
This means it takes a few months before symptoms appear. Certain strains of the parasite can
have a longer incubation period that may last up to a few years.

Who is at risk of echinococcus?

One risk factor for contracting the parasite is exposure to the feces of dogs, cattle, pigs, or
sheep. For example, if you work on a farm with these animals, you may be at higher risk.

Cases of the infection have been reported in the United States, but the risk is higher in
countries where the parasite is more common.

Another risk factor is taking in food or water contaminated with the tapeworm eggs.

What is the outlook for echinococcus?

Your outlook depends on the extent of your infection and the organs affected. In some
instances, cysts may rupture, which can be life threatening. But if your doctor is able to
effectively treat the cysts, your outlook may be good.

How is echinococcus prevented?

There are several different steps you can take to prevent an echinococcus infection. In areas
of the world where the parasite is common, education can help.

Removing the worms from dogs can help stop the spread of infection. Correct disposal of
animal feces can reduce exposure to tapeworm eggs.

Proper handling of cattle at farms and slaughterhouses is also essential. This includes
enforcing meat inspection procedures. Avoiding undercooked or raw beef, pork, and fish can
also help you avoid echinococcus.

Washing fruits and vegetables, especially in areas where the tapeworm is common, may help
prevent infection.

Commonly called dog tape worm, hydatid disease is caused by Ecchinococcus granulosus.
While it is common in the tropics, in the UK, the occasional patient may come from a rural
sheepfarming community. The dog is the defifinitive host and, as a pet, is the commonest
source of infection transmitted to the intermediate hosts – humans, sheep and cattle. In the
dog, the adult worm reaches the small intestine, and the eggs are passed in the faeces. These
eggs are highly resistant to extremes of temperature and may survive for long periods. In the
dog’s intestine, the cyst wall is digested, allowing the protoscolices to develop into adult
worms. Close contact with the infected dog causes contamination by the oral
route, with the ovum thus gaining entry into the human gastrointestinal tract. The cyst is
characterised by three layers, an outer pericyst derived from compressed host organ tissues,
an intermediate hyaline ectocyst which is non-infective and an inner endocyst that is
the germinal membrane and contains viable parasites which can separate forming daughter
cysts. A variant of the disease occurs in colder climates caused by Echinococcus
multilocularis, in which the cyst spreads from the outset by actual invasion rather than
expansion.

Classifification
In 2003, the WHO Informal Working Group on Echinococcosis (WHO-IWGE) proposed a
standardised ultrasound classifification based on the status of activity of the cyst. This is
universally accepted, particularly because it helps to decide on the appropriate management.
Three groups have been recognised:
Group 1: Active group – cysts larger than 2 cm and often fertile.
Group 2: Transition group – cysts starting to degenerate and entering a transitional stage
because of host resistance or treatment, but may contain viable protoscolices.
Group 3: Inactive group – degenerated, partially or totally calci-fified cysts; unlikely to
contain viable protoscolices.
Clinical features
As the parasite can colonise virtually every organ in the body, the condition can be protean in
its presentation. When a sheep farmer, who is otherwise healthy, complains of a gradually
enlarging painful mass in the right upper quadrant with the physical fifindings of a liver
swelling, a hydatid liver cyst should be considered. The liver is the organ most often affected.
The lung is the next most common. The parasite can affect any organ or several organs in the
same patient . The disease may be asymptomatic and discovered coincidentally at post
mortem or when an ultrasound or CT scan is done for some other condition. Symptomatic
disease presents with a swelling causing pressure effects. Thus, a hepatic lesion causes dull
pain from stretching of the liver capsule, and a pulmonary lesion, if large enough, causes
dyspnoea. Daughter cysts may communicate with the biliary tree causing obstructive jaundice
and all the usual clinical features associated with it in addition to
symptoms attributable to a parasitic infestation.Features of raised intracranial pressure or
unexplained headaches in a patient from a sheep-rearing community should raise the sus
picion of a cerebral hydatid cyst.The patient may present as an emergency with severe
abdominal pain following minor trauma when the CT scan may be diagnostic (Fig. 5.17).
Rarely, a patient may present as an emergency with features of anaphylactic shock without
any obvious cause. Such a patient may subsequently cough up white material that contains
scolices that have travelled into the tracheobronchial tree from rupture of a hepatic hydatid on
the diaphragmatic surface of the liver.

There should be a high index of suspicion. Investigations show a raised eosinophil count;
serological tests such as ELISA and immunoelectrophoresis point towards the diagnosis.
Ultrasound and CT scan are the investigations of choice. The CT scan shows a smooth space-
occupying lesion with several septa. An ultrasound of the biliary tract may show abnormality
in the gall bladder and bile ducts. Hydatid infestation of the biliary system should then be
suspected. Ultimately, the diagnosis is made by a combination of good history and clinical
examination supplemented by serology and radiological imaging techniques.

Here, the treatment of hepatic hydatid is outlined as the liver is most commonly affected, but
the same general principles apply whichever organ is involved.These patients should be
treated in a tertiary unit where good teamwork between an expert hepatobiliary surgeon, an
experienced physician and an interventional radiologist is available. Surgical treatment by
minimal access therapy is best summarised by the mnemonic PAIR – puncture, aspiration,
injection and reaspiration. This is done after adequate drug treatment with albendazole,
although praziquantel has also been used, both these drugs being available only on a ‘named
patient’ basis. Whether the patient is treated only medically or in combination with surgery
will depend upon the clinical group (which gives an idea as to its activity), the number of
cysts and their anatomical position. Radical total or partial pericystectomy with omentoplasty
or hepatic segmentectomy (especially if the lesion is in a peripheral part of the liver) are some
of the surgical options. During the operation, scolicidal agents are used, such as hypertonic
saline (15–20%), ethanol (75–95%) or 1% povidone iodine, although some use a 10%
solution. This may cause sclerosing cholangitis if biliary radicles are in communication with
the cyst wall. A laparoscopic approach to these procedures is being tried. Obviously, cysts in
other organs need to be treated in accordance with the actual anatomical site along with the
general principles described. An asymptomatic cyst which is inactive (group
3) may just be observed

Human echinococcosis is a zoonotic disease (a disease that is transmitted to humans

from animals) that is caused by parasites, namely tapeworms of the genus
Echinococcus. Echinococcosis occurs in 4 forms:

cystic echinococcosis, also known as hydatid disease or hydatidosis, caused by infection

with a species complex centred on Echinococcus granulosus;
alveolar echinococcosis, caused by infection with E. multilocularis;
two forms of neotropical echinococcosis: polycystic caused by infection with E. vogeli;
The two most important forms, which are of medical and public health
relevance in humans, are cystic echinococcosis (CE) and alveolar
echinococcosis (AE).

Transmission

A number of herbivorous and omnivorous animals act as intermediate hosts of

Echinococcus. They become infected by ingesting the parasite eggs in contaminated
food and water, and the parasite then develops into larval stages in the viscera.

Carnivores act as definitive hosts for the parasite, and harbour the mature tapeworm
in their intestine. The definitive hosts are infected through the consumption of
viscera of intermediate hosts that contain the parasite larvae.

Humans act as so-called accidental intermediate hosts in the sense that they acquire
infection in the same way as other intermediate hosts, but are not involved in
transmitting the infection to the definitive host.

Several distinct genotypes of E. granulosus are recognised, some having distinct

intermediate host preferences. Some genotypes are considered species distinct from
E. granulosus. Not all genotypes cause infections in humans. The genotype causing
the great majority of cystic echinococcosis infections in humans is principally
maintained in a dog–sheep–dog cycle, yet several other domestic animals may also
be involved, including goats, swine, cattle, camels and yaks.

Alveolar echinococcosis usually occurs in a wildlife cycle between foxes or other

carnivores with small mammals (mostly rodents) acting as intermediate hosts.
Domesticated dogs and cats can also act as definitive hosts.
An adult worm resides in the small intestine of a definitive host. A single gravid
proglottid releases eggs that are passed in the feces of the definitive host. The egg is
theningestedbyanintermediatehost. Theeggthenhatchesinthesmallintestineofthe
intermediate host and releases an oncosphere that penetrates the intestinal wall and moves
through the circulatory system into different organs, in particular the liver and lungs.
Once it has invaded these organs, the oncosphere develops into a cyst. The cyst then
slowly enlarges, creating protoscolices (juvenile scolices), and daughter cysts within the
cyst. The definitive host then becomes infected after ingesting the cyst-containing organs
of the infected intermediate host. After ingestion, the protoscolices attach to the intestine.
They then develop into adult worms and the cycle starts all over again

The adult Echinococcus granulosus (sensu lato) (2—7mm

long) resides in the small intestine of the definitive host. Gravid

proglottids release eggs that are passed in the feces, and are
immediately infectious. After ingestion by a suitable intermediate host, eggs hatch
in the small intestine and release six-hooked
oncospheres that penetrate the intestinal wall and migrate through
the circulatory system into various organs, especially the liver and lungs. In these
organs, the oncosphere develops into a thick-walled hydatid

cyst that enlarges gradually, producing protoscolices and daughter

cysts that fill the cyst interior. The definitive host becomes infected by ingesting the
cyst-containing organs of the infected intermediate host.
After ingestion, the protoscolices evaginate, attach to the intestinal mucosa ,

and develop into adult stages in 32to 80days.

Humans are aberrant intermediate hosts, and become infected by ingesting eggs .

Oncospheres are released in the intestine , and

hydatid cysts develop in a variety of organs . If cysts rupture, the

liberated protoscolices may create secondary cysts in other sites within the body
(secondary echinococcosis).

Alveolar Echinococcosis (Echinococcus multilocularis)

The adult Echinococcus multilocularis (1.2—4.5mm long) resides in the small
intestine of the definitive host. Gravid proglottids release

eggs that are passed in the feces, and are immediately infectious. After ingestion
by a suitable intermediate host, eggs hatch in the small

intestine and releases a six-hooked oncosphere that penetrates the

intestinal wall and migrates through the circulatory system into various organs
(primarily the liver for E. multilocularis). The oncosphere develops into a multi-
chambered (“multilocular”), thin-walled (alveolar) hydatid
cyst that proliferates by successive outward budding. Numerous
protoscolices develop within these cysts. The definitive host becomes infected by
ingesting the cyst-containing organs of the infected
intermediate host. After ingestion, the protoscolices evaginate,

attach to the intestinal mucosa , and develop into adult stages in 32to 80days.
Humans are aberrant intermediate hosts, and become infected by ingesting eggs

. Oncospheres are released in the intestine and

cystsdevelopwithinintheliver . Metastasis or disseminationtoother

organs (e.g., lungs, brain, heart, bone) may occur if protoscolices are released
from cysts, sometimes called “secondary echinococcosis.”

Clinical Presentation

Echinococcusgranulosus infectionsoftenremainasymptomaticforyears before the cysts

grow large enough to cause symptoms in the affected organs. The rate at which
symptoms appear typically depends on the locationofthecyst.
Hepaticandpulmonarysigns/symptomsarethemost common clinical manifestations, as
these are the most common sites for cysts to develop In addition to the liver and lungs,
other organs (spleen, kidneys, heart, bone, and central nervous system, including the
brain and eyes) can also be involved, with resulting symptoms. Rupture of the cysts
can produce a host reaction manifesting as fever, urticaria, eosinophilia, and
potentially anaphylactic shock; rupture of the cyst may also lead to cyst dissemination.

Echinococcus multilocularis affects the liver as a slow growing,

destructivetumor,oftenwithabdominalpainandbiliaryobstructionbeing
the only manifestations evident in early infection. This may be misdiagnosed as
liver cancer. Rarely, metastatic lesions into the lungs, spleen, and brain occur.
Untreated infections have a high fatality rate.

Echinococcus vogeli affects mainly the liver, where it acts as a slow growing tumor;
secondary cystic development is common. Toofewcases of E. oligarthrus have been
reported for characterization of its clinical presentation.

Signs and symptoms

Cystic echinococcosis / hydatid disease

Human infection with E. granulosus leads to the development of one or more

hydatid cysts located most often in the liver and lungs, and less frequently in the
bones, kidneys, spleen, muscles and central nervous system.

The asymptomatic incubation period of the disease can last many years until hydatid
cysts grow to an extent that triggers clinical signs, however approximately half of all
patients that receive medical treatment for infection do so within a few years of their
initial infection with the parasite.

Abdominal pain, nausea and vomiting are commonly seen when hydatids occur in
the liver. If the lung is affected, clinical signs include chronic cough, chest pain and
shortness of breath. Other signs depend on the location of the hydatid cysts and the
pressure exerted on the surrounding tissues. Non-specific signs include anorexia,
weight loss and weakness.

Alveolar echinococcosis

Alveolar echinococcosis is characterized by an asymptomatic incubation period of 5–

15 years and the slow development of a primary tumour-like lesion which is usually
located in the liver. Clinical signs include weight loss, abdominal pain, general
malaise and signs of hepatic failure.

Larval metastases may spread either to organs adjacent to the liver (for example, the
spleen) or distant locations (such as the lungs, or the brain) following dissemination
of the parasite via the blood and lymphatic system. If left untreated, alveolar
echinococcosis is progressive and fatal.
The initial phase of primary infection is always asymptomatic. Many infections are
acquired in childhood but do not cause clinical manifestations until adulthood. Latent
periods of more than 50years before symptoms arise have been reported. While
approximately 50percent of detected cases occur in asymptomatic patients, many
more cases remain undiagnosed or are found incidentally at autopsy.

The clinical presentation of E. granulosus infection depends upon the site of the cysts
and their size. Small and/or calcified cysts may remain asymptomatic indefinitely.
However, symptoms due to mass effect within organs, obstruction of blood or
lymphatic flow, or complications such as rupture or secondary bacterial infections can
result.

Cysts typically increase in diameter at a rate of one to five centimeters per year.
However, cyst growth rates and time courses are highly variable [2,3]. Hydatid cysts
may be found in almost any site of the body, either from primary inoculation or via
secondary spread. The liver is affected in approximately two-thirds of patients, the
lungs in approximately 25percent, and other organs including the brain, muscle,
kidneys, bone, heart, and pancreas in a small proportion of patients. Single-organ
involvement occurs in85to 90percent of patients with E. granulosus infection, and
only one cyst is observed in more than 70 percent of cases

Distribution

Cystic echinococcosis is globally distributed and found in every continent except

Antarctica. Alveolar echinococcosis is confined to the northern hemisphere, in
particular to regions of China, the Russian Federation and countries in
continental Europe and North America.

In endemic regions, human incidence rates for cystic echinococcosis can reach
more than 50 per 100 000 person-years, and prevalence levels as high as 5%–10%
may occur in parts of Argentina, Peru, East Africa, Central Asia and China. In
livestock, the prevalence of cystic echinococcosis found in slaughterhouses in
hyperendemic areas of South America varies from
20%–95% of slaughtered animals.
 The highest prevalence is found in rural areas where older animals are slaughtered.
Depending on the infected species involved, livestock production losses attributable
to cystic echinococcosis result from liver condemnation and may also involve
reduction in carcass weight, decrease in hide value, decrease of milk production, and
reduced fertility.

Diagnosis

Ultrasonography imaging is the technique of choice for the diagnosis of both cystic
echinococcosis and alveolar echinococcosis in humans. This technique is usually
complemented or validated by computed tomography (CT) and/or magnetic
resonance imaging (MRI) scans.

Cysts can be incidentally discovered by radiography. Specific antibodies are

detected by different serological tests and can support the diagnosis.
Early detection of E. granulosus and E. multilocularis infections, especially in low-
resource settings, is still needed to aid in the selection of clinical treatment options.

Your doctor may ask you about your symptoms and perform medical tests to diagnose your
infection. For example, they may use a chest X-ray to rule out other types of infection. Your
doctor may also use an abdominal MRI or CT scan to make their diagnosis.

Because the incubation period can be long, echinococcus parasites may be discovered
while your doctor is performing medical tests for other reasons.

Treatment

Both cystic echinococcosis and alveolar echinococcosis are often expensive and
complicated to treat, sometimes requiring extensive surgery and/or prolonged drug
therapy. There are 4 options for the treatment of cystic echinococcosis:

percutaneous treatment of the hydatid cysts with the PAIR (Puncture,

Aspiration, Injection, Re-aspiration) technique;
surgery
anti-infective drug treatment
“watch and wait”.

The choice must primarily be based on the ultrasound images of the cyst, following a
stage-specific approach, and also on the medical infrastructure and human resources
available.
For alveolar echinococcosis, early diagnosis and radical (tumour-like) surgery
followed by anti-infective prophylaxis with albendazole remain the key elements. If
the lesion is confined, radical surgery can be curative. Unfortunately in many patients
the disease is diagnosed at an advanced stage. As a result, if palliative surgery is
carried out without complete and effective anti-infective treatment, frequent relapses
will occur.

Management approach — Treatment of echinococcosis usually involves antiparasitic

therapy combined with either surgical resection of the cyst or percutaneous
aspiration and instillation of scolicidal agents
Radical procedures include total or partial pericystectomy and hepatic
resections. The cysts are removed completely in radical surgery. A pericystectomy
can be performed using an open or closed cyst method.
Surgical techniques for the treatment of hepatic hydatid cysts range from
hepatic resection to simple cyst evacuation and partial
pericystectomy. These procedures eventually eradicate the entirety of the
parasitic tissue, resulting in complete cure without recurrence

In most cases a ‘conservative’ surgical approach was chosen (group A: 404 cysts).
The main objectives of this treatment were the avoidance of spillage of cyst contents,
the removal of all parasitic elements and the management (if possible), of the residual
pericyst cavity by various methods. Before the evacuation and necessary partial
pericystectomy, the initial steps were decompression of the cysts by needle and trocar,
removal of the parasite and, as a scolicidal procedure, lavage of the cavities with
hypertonic saline swabs. The opening of the pericyst was sutured, or the cavity was
left open, and either obliterated by capitonnage (in folding of the cystic wall by
successive suturing layers) or filled with omentum. In infected cysts
or hepatic cysts with intrabiliary rupture, external drainage of the cavities was usually
undertaken. In cysts of the liver that had ruptured into the biliary tree, the operative
technique was directed towards the cyst, but in addition towards possible suturing of
the cystobiliary communication and drainage of the common bile duct (T tube, two
cases, choledoduodenostomy, one case). The gallbladder was excised, if involved
(nine cases). In
biliobronchial fistula due to thoracic extension of a cyst of the dome of the
liver, besides management of the parasitic cavity, suturing of the pulmonary
side of the fistula and closure of the diaphragmatic defect was the usual
procedure. In calcified cysts of the liver, operated because of their size or
co‐existent infection, pericystectomy was performed with removal of the
calcareous plaques and drainage. In huge calcified cysts a ‘planned’
re‐operation was sometimes carried out with a bloodless chiselling of the
calcification (due to intervening inflammation). In lung hydatids the two
general principles were to remove the parasite and to treat both the
bronchipericyst pathology and associated lesions as conservatively as
possible. The procedure of choice was the tedious delivery of the parasite
by enucleation. Obviously for large cysts, needle aspiration and extraction
 without spillage of the collapsed cysts were essential. Especially important was the
closure of any bronchial openings in the remaining cavities.
In some peritoneal cysts, partial pericystectomy with parasite evacuation and drainage
was selected because of adhesions around the cysts and the danger of bleeding or
rupture of the parasite during the dissection.

A few cases require more radical procedures [1, 4]. In our series cystopericystectomy
was the ideal technique for small calcified hepatic cysts or cysts that were
peripherally located or pedunculated, whenever this time‐consuming method allowed
a plane to be established between the parasite and the host's tissue. Formal resections
were rarely performed for liver and lung cysts because these radical procedures
seemed disproportionate for a benign lesion and because of the possible risk of re‐
infection in the remaining healthy part. In two splenic cysts and one renal cyst
splenectomy and nephrectomy were carried out because of destruction of these
organs.

Some therapeutic principles emerge from these series of multiple cysts

[7, 9, 10]. In multiple cases it is better to manage all the cysts in one stage, adjacent
parasites being easily evacuated through the cavity of the more superficial cyst.
Otherwise, when one of the cysts is in an inaccessible position and the patient's
condition is poor, it is best to plan a two‐stage procedure. In co‐existing liver and
pulmonary parasites the lung cysts should be operated either first, because of the
greater tendency to rupture and infection, or simultaneously (in cysts of the dome of
the liver and right lung). Bilateral lung cysts have to be resected in two stages. In
uncomplicated lung cysts the lung with the larger cyst or with more cysts must be
treated first. In patients with unruptured cysts >5 cm in one lung and a ruptured cyst
in the other, the intact lung has a higher therapeutic priority.

Certain medications can destroy the parasite. In some cases, your doctor may also
recommend surgery. Your specific treatment plan will depend on the severity of your
symptoms, as well as the organs affected.

Medication

Medication is almost always used to treatechinococcus. For example, your doctor may
prescribe mebendazole or albendazole.

They may also recommend taking anti-inflammatory medication to treat inflammation of

your organs caused by the parasite. Sometimes chemotherapy medications can be used to
treat organ cysts caused by the parasite.
Surgery

In some instances, your doctor may recommend surgery to treat cysts caused by the infection.
If the infection has affected your brain and fluid has accumulated there, your doctor may also
recommend surgery to install a shunt. This device is used to drain fluid from your brain.

CAPITONNAGE

The surgical approach to hydatid cyst of the lung is based on complete removal of the
parasite, and management of the residual pericyst cavity. For the latter, capitonnage is a
recognized method, by which the walls of the evacuated pericyst cavity are
approximated with multiple sutures.

Capitonnage without suturing and ligation of bronchial openings individually may lead
to postoperative air leakage with resultant pneumothorax or pneumatocele. The
pulmonary parenchyma may also become distorted. Herein, we report another
complication of capitonnage: presence of large amounts of unabsorbed suture material
acting as foreign body within the residual pericyst cavity, leading to chronic infection
and hemoptysis.

The surgical treatment of hydatid cyst of the lung calls for complete removal of the
parasite and management of the residual pericyst cavity. Rarely, the involved portion of
the lung may be necessarily removed (1).

One of the earliest methods for management of the residual pericyst cavity was
described by an Argentinian surgeon, Posadas in 1890, who introduced simple suture
closure of the pericyst opening. To avoid inevitable air leaks, this technique was
modified by suturing the edges of the pericyst cavity to the thoracotomy incision. In the
same year, Delbet, a French surgeon, proposed folding of the walls of the pericyst cavity
inward by multiple sutures, a technique called capitonnage (2).

In 1946, Ugon et al, according to Crausaz, proposed obliterating the pericyst cavity by a
series of purse-string sutures inserted from the bottom of the pericyst cavity upwards
to the pleural surface (3). This approach was modified in the same year by Allende and
Langer who suggested individual ligation of the bronchial openings. Despite these
refinements, capitonnage continued to remain popular because it could be rapidly
executed (4). All viable hydatid cysts of the lung, in the course of their expansion, cause
pressure necrosis of the walls of adjacent bronchi. The basic problem with capitonnage
is that the bronchial side holes, opening into the pericyst cavity, may be overlooked
once the parasite has been removed. The walls of these bronchial openings, if larger
than 1-2 millimeters, are usually cartilaginous and cannot be completely sealed off by
merely approximating opposing surfaces of the pericyst cavity. The stage is set for
persistent postoperative air leakage. The other drawback of capitonnage is that it
distorts the pulmonary parenchyma and leads to atelectasis especially when there are
multiple cysts (5).

We encountered another problem with capitonnage: a large amount of unabsorbed

suture material remained in the pericyst cavity, acting as a foreign body and
perpetuating the disease process.

CASE SUMMARIES

A 20 year-old man presented with a history of single hydatid cyst of the right lower lobe
removed 14 months earlier. He was readmitted because of intermittent hemoptysis, raising
the possibility of local recurrence of the cyst. A chest CT scan (Figure 1) further increased
this suspicion. Bronchoscopy showed fresh blood clots in the right lower lobe bronchus.
During thoracotomy, an irregular cavity was found in the right lower lobe just beneath the
major fissure, measuring seven centimeters in diameter. The cavity contained frank pus, two
large knotted 2-0 monofilament nylon suture segments and no hydatid material. More than
six separate bronchial side holes opened into the irregular linings of the pericyst cavity, two
of them larger than 4 mm in diameter. The suture material was removed and all bronchial
openings were closed individually with 3-0 nylon sutures. The pericyst cavity was left open
to the pleural surface and the edges of the opening were overrun with 3-0 chromic catgut
sutures. The postoperative course was uncomplicated.

DISCUSSION

There is no surgical dictum stating that residual pericyst cavities of hydatid cysts must
be closed or somehow obliterated (6). The basic surgical technique demands careful
closure of bronchial openings regardless of their size. Capitonnage alone, by simply
bringing two surfaces of the pericyst lining into apposition, cannot ensure complete and
lasting closure of bronchial openings. A pneumatocele may form if the site of entry into
the pleural cavity is closed, or a pneumothorax may occur if it has been left open. For
very large pericyst cavities, many sutures may be required to fully close the pericyst
cavity and they may act as foreign body if remain unabsorbed. After removal of the
hydatid cyst of the lung, it is essential to take the time and effort to identify all the
bronchial openings within the pericyst cavity. They should be individually closed with
fine non-absorbable sutures. There is no need to close the residual pericyst cavity, as it
will become rapidly repleuralized and the lung will fully expand.

As a final maneuver, the edges of the residual pericyst cavity should be overrun with
continuous absorbable sutures to avoid minor bleeding and air leakage. Another
advantage of this approach is that a pericyst cavity left open to the pleural space allows
for early detection of inadvertently missed bronchial openings by observing continuous
escape of air via the chest tube. This can lead to early intervention, before devastating
complications like empyema occur.
Surgery of uncomplicated pulmonary hydatid cysts: Capitonnage or uncapitonnage?

Abstract

Purpose
The aim of this study is to compare two methods of capitonnage and uncapitonnage
in the surgery of uncomplicated pulmonary hydatid cyst

We conclude that the capitonnage method has more advantages in surgery of

uncomplicated pulmonary hydatid cysts.

Hydatid cyst
Lung
Surgery
Capitonnage

1. Introduction
Hydatid disease is an important public health problem in Iran.1 It is common in the
rural areas where cattle and dogs are in abundance. The primary hosts of
the Echinococcous granulosis tapeworms are dogs and canine. These tapeworms
produce eggs in the intestine and pass them via stool. Eggs are ingested by
intermediate hosts such as sheep and cows. Human is considered accidental host by
digesting tapeworm eggs.2Surgical methods for pulmonary hydatid cysts are
enucleation or puncture and evacuation of cyst contents and Cystotomy with or
without capitonnage. There is controversy over advantages of capitonnage and
uncapitonnage method in the treatment of pulmonary hydatid cyst. In the present
retrospective comparative study, the advantages of two above mentioned surgical
methods in the surgery of uncomplicated pulmonary hydatid cysts have been studied.

2. Patients and methods

This retrospective study was carried out on hospitalized patients who were operated
for pulmonary hydatid cyst in Imam Reza Medical Center, Tabriz, Iran from 1994 to
2007. The study was approved by the Medical Ethics Committee of Tabriz University
of Medical Sciences. Of 502 patients, 62 (14%) patients were in pediatric age group
(<12 years old with a mean age of 10.5 years ranging from 3 to 12 years) and 440
(87.5%) patients were adolescents and adults

2 Operative techniques of capitonnage and uncapitonnage method in

uncomplicated pulmonary hydatid cyst
Standard classic posterolateral and anterolateral thoracotomy were used in
uncomplicated pulmonary hydatid cyst of both groups. All patients underwent
general anesthesia. Double lumen tube (DLT) was used in order to avoid the possible
spillage of cyst contents into the contra lateral bronchus and correct position was
confirmed by fiberoptic bronchoscopy. In the anterior and middle lobe cysts,
anterolateral thoracotomy incisions were selected. When cysts were located in
posterior and upper lobes of the lung, posterolateral thoracotomy incisions were
used. We did not perform the surgery on concurrent bilateral intact pulmonary
hydatid cysts. After entering the hemithorax, the lung was spared from adhesions of
chest wall and then cysts were identified and surrounded by 5% povidone iodine
irrigated abdominal lap to prevent seeding of possible ruptured laminated
membrane. Pericyst wall was opened and the laminated membrane was delivered
and enucleated while positive intrapulmonary pressure was applied. With this
maneuver air escaped through bronchial openings and separated the laminated
membrane. Aspiration and evacuation of intact cysts were performed on five giant
hydatid cysts (greater than 12 cm).
Bronchial openings were identified using intrapulmonary positive pressure
maneuver and irrigation of saline into the cystic cavity and closed by 3/0 polyglactin
(Coated vicryl, Ethicon). The cavities were left open after closure of the bronchial
openings in uncapitonnage method. In capitonnage method, all of the cavities were
obliterated by imbrications sutures from deep to superficial layers using the same
suture material. The parts of the pericyst that protruded outside the lung
parenchyma were excised. Minor parenchmal air leaks were also sutured. After
insertion of two chest tubes 30 or 32 French sizes in the pleural cavity, one in the
posterolateral and the other in the upper pleural spaces to drain blood and air, the
chest was closed using classic approach.
There was no selection bias regarding choosing appropriate method including
capitonnage or uncapitonnage for either small or big cysts in this study.
All patients received prophylactic antibiotic therapy with cephazolin 30 mg/Kg/day
perioperatively. During postoperative period chest tubes were removed when no air
leak was evident.
Three to seven days is accepted duration to remove the chest tubes routinely.
Decisions for reoperation and second thoracotomy were made after 14 days or when
massive air leak was present in chest tubes that were considered as bronchopleural
fistula.
Two of our Thoracic surgeons preferred to use capitonnage and two others preferred
to use uncapitonnage method in uncomplicated pulmonary hydatid cyst without any
reason.
After discharge, in the follow up period, chest X-rays were taken for all the patients.
CT scan of the chest was indicated when a suspicious lesion on chest X-ray appeared.
The impact of surgical results was assessed by total hospitalization, duration of air
leak from chest tubes, occurrence of bronchopleural fistula, prolonged air leak
duration, empyema and recurrence.
To control air leak complication and pneumothorax, the primary thoracotomy
incisions were used.
All patients were followed up in surgical outpatient clinic and all relevant
information was recorded in their files for at least two years.
3. Statistical analysis
Descriptive analyses of variables were used to summarize data and the independent
samples t-test was used to compare continuous variables. Chi square or Fisher’s
Exact test was used to compare quantitative variables. SPSS for windows version 16.0
was used to analyze the data. P value less than 0.05 was considered significant.

4. Discussion
Hydatid disease is a major health problem in Northwest of Iran and it is considered
as an endemic disease in this area.1 In most patients with uncomplicated pulmonary
hydatid cyst, lung cysts are incidental findings. Some patients suffer cough, dyspnea,
and chest pain. Complicated hydatid cysts tend to have more severed
symptoms.3 Although medical therapy is suggested to be effective in some reports by
degeneration changes of laminated membrane, surgery is still the mostly accepted
treatment of pulmonary hydatid cyst.4 The cure rates for medical treatment are only
25%–30%. Medical treatment (Albendazole) is indicated in ruptured hydatid cysts or
in preventing recurrences after surgery.5, 6 Delbet is the first one who described the
capitonnage method in the surgical treatment of pulmonary hydatid cyst.7
There was controversy over doing the surgical methods of pulmonary hydatid cyst.
Some reports of the literature are as following: Saidi’s work showed that after closure
of bronchial opening, approximation of suturing cavity is not necessary because the
cavity space was obliterated later by pulmonary parenchyma and pleura.8 Turna
studied 71 patients of pulmonary hydatid cyst. From which 34 patients had intact and
37 patients had complicated pulmonary hydatid cyst. Turna concluded that
capitonnage approach had no advantage in the surgery of both complicated and
uncomplicated forms of pulmonary hydatid cysts.9 Nessimi and associates studied
113 patients with both forms of pulmonary hydatid cysts. 33 patients of their series
were operated by uncapitonnage and the remaining with capitonnage method. They
confirmed that noncapitonnage method patients had shorter mean hospital stay with
higher morbidity than capitonnage method patients and noncapitonnage method
may be a good alternative to the capitonnage procedure.10 Sonmez and associates
reported that in 15 patients, eight patients were treated surgically with capitonnage
and seven patients with uncapitonnage methods in lung hydatid cyst regarding to
hospital stay and postoperative chest tube removal days. They concluded that
capitonnage method had shortened postoperative air leak and chest tube drainage
removal times and reduced morbidity in comparison with uncapitonnage
method.11 In 60 patients of Kosar’s series in complicated and uncomplicated forms
of pulmonary hydatid cysts, 37 patients were treated surgically by capitonnage and
23 patients with uncapitonnage methods and concluded that capitonnage method for
pulmonary hydatid cysts is superior to Cystotomy and reduces morbidity (especially
prolonged air leak from chest tubes and hospital stay).12 Erdogan and associates also
studied the surgery of both forms of complicated and intact pulmonary hydatid cysts
in 89 patients, 44 patients were treated with capitonnage and 45 patients with
uncapitonnage methods and concluded that capitonnage procedure was not
advantageous.13
Their works are comparing the advantages of uncapitonnage or capitonnage method
in complicated and uncomplicated forms of pulmonary hydatid cysts.
Only did Biglin and associates study the surgery of intact pulmonary hydatid cyst in
the literature. This study was carried out on 59 patients with 70 cysts (32 patients
with capitonnage and 27 patients with uncapitonnage methods). The result of this
randomized clinical trial study showed that capitonnage method is a procedure with
low complication rates and is a preferred approach in intact pulmonary hydatid
cysts.14 Our study is similar to Biglin’s, and approves the advantages of capitonnage
surgical method in uncomplicated pulmonary hydatid cysts.
Pulmonary resection for intact hydatid cyst is rarely required.14
In one study, Albendazole therapy has been recommended 7–10 days before and one
month after surgery in intact hydatid cysts.15 In our study no preoperative medical
therapy was used routinely and most patients received medical treatment with
Mendazole or Albendazole after surgery for 1–3 months.
Incidence of recurrences of the disease in both groups of our study was low because
most patients were operated using enucleation and delivery procedures as part of the
surgical method. Two cases of giant cyst did not recur though spillage and
contamination of cyst cavity with scolicis occurred during surgery. This approves also
the efficacy of Albendazole in preventing recurrence.
The incidences of recurrences of both groups in our study were not significant.
Symbas and Aletras showed that larger cysts are associated with an increased risk of
rupture with enucleation method.16 Enucleation method was not used in the surgery
of our giant cysts.
Advantages of uncapitonnage method in complicated hydatid cysts reported by
Turna, Nessimi and Erdoghan may be due to the presence of infection and
inflammation of adjacent pulmonary tissues that made cystic cavity be left open.
Capitonnage sutures in complicated patients can lead to infection and laceration of
the pulmonary tissue and disruption of sutures.
In our and Biglin’s studies the cysts were intact and there was no infection,
inflammation or microbial contamination. So we think, in intact pulmonary hydatid
cysts after closure of bronchial openings, we can close cyst cavities by imbrications
sutures without any remarkable postoperative complications.
In most studies as our study no mortality was reported.
Video assisted thoracoscopy surgery is suggested with less reliability. It is possible to
remove the ruptured laminated membrane to pleural spaces, but spillage of cyst
content may cause severe complications as anaphylaxis, or pleural bacterial
spread.17 We believe that open surgery is the ideal method for pulmonary hydatid
cyst. None of our patients were operated with thoracoscopic approach.
The radiologic appearances of uncomplicated pulmonary hydatid cyst found in our
series were as following: Coin and notch sign5; moon or crescent
sign.18 Ultrasonography of thorax and abdomen are useful for diagnosis of
pulmonary hydatidosis.19 However, it was used in 50% of our cases, so the data
related to ultrasound findings have not been used in the present study.
The current study has some limitations as following
The methodological problem generally found in applying. This method (capitonnage)
has been discussed to have advantage in intact pulmonary hydatid cysts in one
report.14 However, further experimental validation is necessary
Medical treatment (Mebendazole) with surgery was considered for patients from
1994 to 1999. This medical treatment was changed during 2000–2007 with
Albendazole. Since this study started in 1994, we used older medical treatment which
needs to be changed in future studies.
This study is not blinded.

Our results, however may gain wider acceptability if verified through more extensive
studies with multicenter and larger sample series to confirm our results.

5. Conclusion
Our capitonnage series had surprisingly higher and better results than
uncapitonnage approaches. Capitonnage approach for uncomplicated pulmonary
hydatid cysts is superior to uncapitonnage method because it reduces complications,
morbidity and rate of reoperation

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