Mohammad

Sultan

Khuroo,

MD,

Echinococcu.s Management

hepatic Echinococcus cysts (maximal diameter, ± 4.0) in 12 patients were aspi-

7.5 cm rated and (20%)

irrigated

saline

with

under

the

cysts,

which

hypertonic

sonographic

ance. All patients had symptoms of a hepatic by

guid-

signs mass

had

and caused

a prominent

of scoleces were observed Mean hospital stay was ± 3.4. Serial sonographic

days nations revealed high-level the cyst cavity (heterogeneous pattern), uniformly

which

ultimately

in all 4.0 exami-

echoes in echo became

echogenic (pseudotumor). After follow-up of 14.0 months ± 5.5, maximal cyst diameter decreased to 4.1 cm ± 3.1 (P < .001). One patient died of unrelated causes; the remaining 11 patients experienced relief of symptoms and a decrease in liver span. Index terms: Cyst, percutaneous drainage, 761.3121 #{149} Echinococcosis, 761.2083 #{149} Liver, cysts, 761.3121 #{149} Liver, echinococcosis, 761.2083 #{149} Liver, interventional procedure, 761.12986 #{149} Liver,USstudies,761.12986 #{149} Parasites, 76.2083 Radiology

1991;

Ali Zargar,

H

YDATID

man

and larval

180:141-145

(2).

to the

of the

cases

0 RSNA,

1991

Received

in

primary

cysts,

of disseminated or disease is a much (3). Medical therapy

has

especially

advantages,

October

15, 1990;

re11.

in patients

with recurrent disease or in patients for whom surgery is otherwise madvisable (1,3-5). There are some questions about its efficacy and safety (57). Percutaneous puncture or aspiration of hydatid cysts has been contraindicated (8). However, there are reports

of hydatid

cysts

being

punc-

tured and neither anaphylaxis nor peritoneal soilage resulting (2,9,10). In this study, we report the results of ultrasound (US)-guided percutaneous

transhepatic

patic

hydatid

drainage

cysts

MATERIALS

of 21 he-

in 12 patients.

AND

METHODS

From June 1988 onward, all patients with hydatid disease of the liver being treated at our institution were considered for treatment with percutaneous drainage

(a)

November 15; revision 26, 1991; accepted March requests to M.S.K.

re-

successful

but management recurrent hydatid greater problem

symptoms caused by

cyst

with

that

appeared

wall

in Table

fluid

anechoic

a

or hypoechoic,

primary

echoes,

form

excluded solid

of treat-

if the pattern

(b) was

cyst with-

infected,

The

cysts,

clinical

formed

parameters

the

study

of the

intradermal

1gM antibodies (measured

test

of immunogbobulin

to

G

Echinococcus

by means

of the en-

zyme-linked immunosorbent assay [ELISA]) were performed and the results evaluated, as reported earlier (12). The patient fasted overnight. The procedure

group.

group

2. The Casoni

measurement

was

performed

the

next

morning,

or

had ruptured into the biliary tree or pleural or peritoneal cavity. Twelve patients, harboring 21 hepatic hydatid

and

(IgG) and granulosus

component

enhancement of back wall (c) gave informed consent to

Patients were had a hyperechoic

out back

shown in Table 1. The types and descriptions of the hydatid cysts (11) are shown

if the patient

and signs of a hepatic a hydatid cyst; (b) had

a prominent

(a) (c) ment.

India.

MD

the

surgical

cysts-is

echoes; and use of PD as the

requested February reprint

Mahajan,

In the Liver: Drainage’

bordering

of uncomplicated

with marked

(Kashmir),

areas

Treatment-usually

moval

had

revision ceived Address

#{149} Rakesh

Cysts

(PD). PD was performed

the Department of Gastroenterology, of Medical Sciences, Srinagar 190 011

DM

Mediterranean and Baltic seas, South America, Australia, the Middle East, and New Zealand. Immigration has bed to an increased prevalence of the disease in Europe and North America

mass

From Institute

MD,

disease is the commonest one of the most severe hucestodiases (1). The disease

is endemic

fluid component that appeared anechoic or hypoechoic, with marked enhancement of back wall echoes. The amounts of cyst fluid aspirated and of hypertonic saline used were 190 mL ± 240 and 120 mL ± 90, respectively. Separation of the endocyst from the pericyst and nonviability cysts.

#{149} Showkat

granulosus with Percutaneous

Twenty-one granulosus

DM

are

Abbreviations: ELISA = enzyme-linked munosorbent assay, Ig = immunoglobulin, PD = percutaneous drainage.

im-

141

Table

2

Table

Distribution

of 21 Cysts into Five

According et al (11)

Types

Gharbi

3

of Cyst Fluid Aspirated

Giaracterisfics

to Classification

of

before

and after Infusion

of Saline

into Cyst

Cavity No. of Cysts

with

Characteristic

No. of Description

Type

Before

Cysts

of

Characteristic

I

Pure fluid collection;

ifi

Iv

V

After Infusion

of

Saline

14 Appearance

rounded with welldefined borders Fluid collection with split wall (localized) Fluid collection with multiple septa; honeycomb appearance Hypoechoic with high internal echoes Cyst with reflecting

H

Infusion Saline

and watery

Clear

0

20

Opalescent

1

0 1

Turbid

21 0

Culture

3

Negative

19

Positive

Cytologic

2

thick walls *

t

Escherichia

13 (12) 4

21 (1)* 7 3

1

coil (one

patient)

Staphylococcusepidermidis

S Numbers

1

findings

Scoleces Hookiets Membrane

1

20

2*

and (skin

in parentheses

Salmonella

typhimurium

(one

patient).

contaminant).

are number

of cysts

with

scoleces.

viable

with close monitoring. Facilities were available to treat any potential complication. The location of the cyst was defined in three

planes

with

sonography

by using

a real-time linear scanner (SSD 256; Aloka, Tokyo) with a 3.5-MHz probe. The relation of the cyst to the normal liver was delineated, and a site for puncture was marked such that the cyst could be approached through thick normal liver tissue. If possi-

visit,

patients

underwent

clinical

examina-

patients masses,

tion and had a blood sample drawn for serum chemistry and serologic testing. Sonography

was

performed

to PD, were examination.

to evaluate

the diameter and appearance of the cyst. All values were expressed as means ± one

ble, the right intercostal route was preferred to minimize the chances of peritoneal soilage. Under aseptic conditions, USguided cyst puncture was performed through the biopsy port of the puncture probe (3.5 MHz). A transhepatic catheter needle (5-F, 40-cm-long radiopaque polyethylene catheter; Cook Europe, Bjaerver-

standard

skov,

All of the 21 cysts in the 12 patients were successfully treated with PD. The transhepatic catheter needle was used to drain 13 cysts, while the cholangiographic needle was used to drain eight cysts. The amounts of cyst fluid aspirated and of saline infused into the cyst cavity were 190 mL ± 240 (range, 2.5-600 mL) and 120 mL ± 90 (range, 2-450 mL), respectively. The characteristics of the cyst fluid obtained before and after infusion of saline into the cyst cavity are shown in Table 3. In one patient, scoleces were viable in the cyst fluid aspirated after infusion of saline. PD was repeated, and the scoleces became nonviable. The mean hospital stay was 4.0

Denmark)

was

tion of unilocular cysts

containing

giography

employed

for aspira-

large-volume daughter

needle

(22

cysts.

cysts,

gauge,

In

a cholan-

20 cm

bong

Cook Europe) was used to puncture each daughter cyst; individual daughter cysts were then aspirated and irrigated with hypertonic saline. In small-volume cysts, the cholangiography needle was the preferred

choice.

puncture,

Immediately

after

cyst

cyst fluid was aspirated. was

sterile ration

hypertonic (20%) saline. Cyst aspiand reffiling was monitored contin-

uously

ifiled

by means

with

an equal

The

cavity

volume

of sonography.

of

The

hy-

pertonic saline was left in the cyst for 20 minutes, after which the fluid was aspirated. At this time, separation of the endocyst

from

reduce

exudation

cavity

the pericyst

was

Cyst

with

filled with

fluid

observed.

To

from the cyst wall, the

washed

left partly

was

was

normal

saline

and

to cytologic

and microbiologic examination. For cytologic examination, the fluid was centrifuged and the sediment examined for fragments hooklets,

of the laminated and scoleces. The

scoleces

was

motility

at immediate

microscopy

staining

with

eosin

tient

assessed

membrane, viability

neutral

was observed

charged

from

by observing

for 48 hours

the hospital

#{149} Radiology

their

and

(13). The

pa-

and dis-

if the procedure

had been uneventful. Thereafter, were followed up every month. 142

of

patients At each

The

in statistical

paired

not

palpable

Cystic prior

at follow-up

Student

analysis

of the

Cyst

Size

and

Appearance

results.

Repeated tions

RESULTS PD

days

Outcome

±

3.4 (range,

patients left the hours after PD.

solution.

subjected

deviation.

test was used

with hepatomegaly. palpable in six patients

4-12

hospital

days).

within

Nine

5.5 (range,

and

pearance diameter 4.1

cm

size

patients

cysts)

were

(P


1:160) had a fourfold drop in absorbance and a negative serologic titer. None of the patients had a rise in the hydatid antibody titer, either of the IgG or the 1gM type.

cyst

in eight examined

contents were studied ± 1.5 (range, 1-12 weeks)

10.3 after

weeks PD. The fluid was turbid in all 14 cysts, and bile stained in three cysts. Cultures for both aerobic and anaerobic

microorganisms

and

microscopy

were

bris, and ples.

hydatid membranes, hooklets, dead scobeces in all the fluid

revealed

negative,

cellular

desam-

Complications Cyst

Reaspiration

We were term Volume

viability 180

During

concerned

about

of the

cyst

Number

1

#{149}

long-

contents

af-

PD

and

the

low-up period, none developed anaphylaxis,

immediate

fol-

of the patients asthma, or

Note and

laryngeal edema. type I) developed after the procedure. lasted

for

48 hours,

multiple high-level

septa and internal

One patient (cyst urticaria 6 hours The urticaria and

the

patient

responded to antihistaminic therapy. Two patients (cyst types I and V) developed fever within 48 hours after PD. Cyst fluid from both patients showed growth of microorganisms (E coli in one and S typhimurium in the other). Specific antibiotic therapy was instituted, resulting in clinical recovery.

One patient (cyst type II) developed biliary rupture of a hydatid cyst 4 weeks after PD. At endoscopic retrograde cholangiopancreatography, a wide endoscopic sphincterotomy was performed and laminated membranes were basketed out of the bile duct. At Radiology

143

#{149}

serial

sonographic

cyst

cavity

examinations,

was

replaced

by

the an

echogenic mass that eventually lost its rounded shape and was not visible at repeated sonography. One patient with six liver cysts died of massive hemoptysis 3 months after PD. Autopsy revealed a mediastinal tubercular gland eroding into the pubmonary vein and bronchial tree.

and

cyst

bly

risks

puncture has been due to the potential

of anaphylactic

shock,

peritoneal

seeding and dissemination, and growth of secondary peritoneab cysts. Although anaphylactic shock from rupture of an echinococcal cyst has been documented, its exact frequency and mechanism have not been well studied (9). Lewall and McCorkell studied 20 patients with rupture of echinococcal cysts in the liver; none developed anaphylaxis (14). Schiller studied complications of echinococcal cyst

rupture

in 30 patients.

tients had tachypnea, and hypotension and hours of cyst rupture; patients had generalized autopsy

(15).

These

Two

pa-

tachycardia, died within 48 both of these peritonitis at authors

con-

cluded that an anaphylactic reaction occurs infrequently following cyst rupture and spillage. Percutaneous catheter drainage is the treatment of choice for the majority

of intraabdominal

regardless no evidence ity

contents

ous

occurs

aspiration

abscesses

fluid

of etiology that any

not

during

of fluid

(16,18).

The

There is of cay-

percutane-

collections use

of fine

and nee-

dles and catheters; advances in radiographic, sonographic, and CT techniques; an approach through thick liver tissue; a preference for the right intercostal approach; and sudden complete decompression immediately after puncture make the chance of spillage extremely low and perhaps less

than

what

might

occur

with

sur-

gical manipulation. PD was performed with close monitoring and with facilities available for treating any potential complication, especially anaphylaxis. Hydatid cyst aspiration and/or drainage has been performed and reported in the literature (2,9,10,19). Of the 15 patients whom we are aware of in whom hydatid cysts were aspirated, none deveboped anaphylaxis. In the present study, 21 cysts were aspirated in 12 patients, none of whom developed asthma,

laxis. 144

laryngeal

Two

patients

#{149} Radiology

related

edema,

developed

or anaphy-

fever,

to PD.

This

findings contained

infu-

patient

had

eventually

are caused by rupture (14).

can

lead

rupture due to high sure. Two earlier reports therapeutic drainage cysts

in the

reported

pigtail cavity

of a patient with six hepatic hydatid cysts. Scan was obtained 15 days after four cysts were drained in a single session. Note separated endocyst in four cysts and intact endocyst in two cysts. Arrows indicate sites of cysts.

to biliary

intracystic

pres-

have described of echinococcal

(2,9).

Mueller

patient

who

et ab (9)

underwent

PD of a recurrent hydatid liver. In their case, a 8.3-F

catheter was for 3 months.

tinuous ity

liver

one

successful cyst of the

catheter

left in place This allowed

drainage

for a prolonged

(2) treated three cysts of the liver

in the con-

of the

cay-

Bret

et al

period.

stage.

We

datid

cysts

tinuous endocyst

all of the

in a single

With

catheter drainage, membrane would

quently blocked the and helped introduce residual

cavity.

catheter lumen infection into Also,

have

been

impractical

ters

in small

cyst

cavities.

However,

the

cyst

ducts and/or endoscopic

cyst

from

contents

the

common

common

bile

duct

may

function: agent

it

and

the endocyst

also

from

pattern;

months

± 5.5

the

follow-up

(range,

3-18

of 14.0 months)

in

insufficient to clinical out-

To conclusively we

are

follow-up

answer

this

performing

of the

long-

patients

in this

study.

However,

the

earliest

response

to cyst

regrowth

or dissemination

is

tive in all three patients at the last follow-up. None of the 12 patients had a rise in 1gM antibody titer at serial follow-up. We believe that the period of follow-up was sufficient for 1gM antibody levels to rise if peritoneal dissemination and cyst growth had occurred. Serial sonographic examination of

of

bile

at endoscopic

retrograde cholangiopancreatography in one patient. Such patients

to

the appearance of an 1gM-type antibody response to E granulosus (12). All three of our patients with a positive 1gM ELISA titer showed a fall in the antibody titers, and the test was nega-

duct has been performed (20). In the present study, we were successful in removing laminated membrane from the

growth

term

contents

gablbladremoval

separate

question,

meticulous evacuation of cyst contents and surgery. Cysts that communicate with the biliary tree require to evacuate

(20%)

the pericyst. This agent is effective and safe, has no systemic toxic effects, and does not cause secondary sclerosing chobangitis when bile duct communication is present (21). E granulosus cysts have a slow

tion?

successful in cysts with a hyperechoic solid pattern without back wall shadows. Infected hydatid cysts require

from the bile der. However,

and

come of PD. Can cyst growth occur locally or in the peritoneal cavity due to cyst fluid soilage and dissemina-

cathe-

patients with suspected bibiary communication might benefit from prolonged cyst drainage, as did the patient reported by Mueller et al. Such cavities continue to drain for a long time; in many there is frank biliary rupture of cyst material, as occurred in one of our patients. We performed PD in hydatid cysts with a prominent fluid component. The procedure would not have been

surgery

a dual

the present study was evaluate the long-term

it would

to place

saline

as a scolexcidal

helped

con-

hypertonic

performed

acted

the thick have fre-

of PD

the cysts. The saline, which left in the cyst cavity for 20 mm-

utes,

21 hy-

stage.

We used was

patients with hydatid with PD. After aspi-

drained

benefit from a combination endoscopic basketing. irrigate

ration, scolexcidab irrigation, and reaspiration of the cyst cavity, the catheter was immediately removed and the cyst was thus treated in a single

the

collections,

(16,17). spillage

before

a cyst with a split wall at US before the procedure was performed. Such

This

cyst

aspirated

showed growth of miThis led us to believe

that the cysts were already harboring bacteria, which were reactivated by aspiration. Similarly, biliary rupture of a type II cyst in one patient was possi-

sonographic cysts with

DISCUSSION Hydatid contraindicated

fluid

sion of saline croorganisms.

all of the

treated

changes. high-level

The cavities were echoes consisting

bar debris, dead and membranes. slowly

solidified

further

fluid

docyst,

and

cysts

revealed

scoleces, These

hooklets, contents

owing

secretion the

cyst

similar

filled with of cellu-

to the

by the gave

the

lack

of

enappearJuly

1991

ance of a pseudotumor. Similar sonographic appearances have been reported by other researchers after PD

(2,8)

long-term Until treatment cysts

and

in patients

receiving

albendazole therapy (4). now, surgery has been the of choice for E granulosus

(21).

Surgery

of uncomplicated

sterile,

unilocular cysts is effective, with a morbidity of approximately 8% and a mean hospital stay of 11.8 days. The surgical morbidity increases-as does the hospital stay-in cases in which cyst drainage is performed; in patients

with

diseases;

cardiac

and

cessible

and

in cases

sites,

multiple

pulmonary

of cysts

may

and

have

fatal

at mac-

disseminated

those recurring after surgical procedures. Peritoneal age is known to occur at surgery cysts,

consequences;

prior spilland 50%

of

surviving patients continue to harbor the disease (21). PD-as assessed in 12 patients in the present study-was effective and safe and resulted in a much shorter hospital stay (4.0 days ± 3.4). PD has a particular advantage over surgery, as it can be performed in patients whose cysts are inaccessible to surgery or who are high surgical risks due to systemic illnesses. PD is particularly safe in patients with recurrence of cysts after surgery,

as the

aspirating

needle

can

be directed through the scar tissue to reduce the chances of cyst leakage and peritoneal soilage. Albendazole, a benzimidazole, has been shown to be effective in human hydatid disease (1,3-5). The drug produces high concentrations of albendazobe sulfoxide in the blood, cyst fluid, and cyst wall (1). It causes death of scoleces, and cysts show objective evidence of reduction in size and may even disappear (5). However, pro-

Volume

180

#{149} Number

1

longed therapy is needed, the drug is teratogenic and hepatotoxic, and some patients fail to respond (4-7). PD can be supplemented with albendazole therapy in a number of ways to achieve better results: abbendazole can be used before PD to make cysts albendazobe

sulfoxide

can

ud-Din,

The

PA, for secretarial

authors

thank

9.

10.

be

introduced into the cyst cavity as a scobexcidab agent, and PD can be foblowed by albendazole therapy to kill any live scoleces in the cyst or in the peritoneum. We believe that, in the future, management of most hydatid cysts of the liver will be possible with a combination of drug therapy and PD. Surgery will be needed for those cysts that defy this combination treatment. U Acknowledgment

8.

11.

12.

13.

14.

Mehraj-

assistance. 15.

References 1.

2.

3.

4.

5.

6.

Saimot AG, Meulemans A, Cremieux AC, et al. Albendazole as a potential treatment for human hydatidosis. Lancet 1983; 2:652656. Bret PM, Fond A, Bretagnolle M, et al. Percutaneous aspiration and drainage of hydatid cysts in the liver. Radiology 1988; 168:617-620. Bezzi M, Teggi A, Rosa FD, et al. Abdominal hydatid disease: US findings during medical treatment. Radiology 1987; 162:9195. Morris DL, Dykes PW, Dickson B, Marriner SE, BoganjA, Burrows FGO. Albendazole in hydatid disease. Br Med J 1983; 286:103104. Morris DL, Dykes PW, Marriner 5, et al. Albendazole: objective evidence of response in human hydatid disease. JAMA 1985; 253:2053-2057. Gil-Grande LA, Boixeda D, Garcia-Hoz F, et

al.

7.

Treatment

of liver

hydatid

disease

with mebendazole: a prospective study of 13 cases. Am J Gastroenterol 1983; 78:584588. Schantz PM, Van den Bossche H, EckertJ. Chemotherapy for larval echinococcosis in animals and humans: report of a workshop. Z Parasitenkd 1982; 67:5-26.

16.

17.

18. 19.

20.

Lewis

JW,

Koss

N,

Kerstein

MD.

A review

of echinococcal disease. Ann Surg 1975; 181:390-396. Mueller PR, Dawson SL, Ferrucci JTJr, Nardi GL. Hepatic echinococcal cyst: successful percutaneous drainage. Radiology 1985; 155:627-628. McCorkell

SJ.

Unintended

percutaneous

aspiration of pulmonary echinococcal cyst. AJR 1984; 143:123-126. Gharbi HA, Hassine W, Brauner MW, Dupuch K. Ultrasound examination of the hydatid liver. Radiology 1981; 139:459-463. Wattal C, Malla N, Khan 5, Agarwal C. Comparative evaluation of enzyme-linked immunosorbent assay for the diagnosis of pulmonary echinococcosis. J Clin Microbiol 1986; 24:41-46. Smyth JD, Barrett NS. Procedures for testing the viability of human hydatid cysts following surgical removal, especially after chemotherapy. Trans R Soc Trop Med Hyg 1980; 74:649-652. Lewall DB, McCorkell SJ. Rupture of echinococcal cysts: diagnosis, classification, and clinical implications. AJR 1986; 146: 391-394. Schiller CF. Complications of Echinococcus cyst rupture: a study of 30 cases. JAMA 1966; 195:220-222. vanSonnenberg E, Mueller PR, FerrucciJT Jr. Percutaneous drainage of 250 abdominal abscesses and fluid collections. I. Results, features, and complications. Radiology 1984; 151:337-341. Mueller PR, vanSonnenberg E, Ferrucci JT Jr. Percutaneous drainage of 250 abdominal abscesses in fluid collections. II. Current procedural concepts. Radiology 1984; 151: 343-347. Welch CE, Malt RA. Abdominal surgery. N EngI J Med 1983; 308:753-760. Livraghi T, Bosoni A, Giordano F, Lai N, Vettori C. Diagnosis of hydatid cyst by percutaneous aspiration: value of electrolyte determinations. JCU 1985; 13:333-337. Karawi

MA,

Hanid

MA.

Endoscopic

moval of daughter Echinococcus cysts the common bile duct. Hepatogastroenterology 1985; 32:296-298. 21.

LangerJC,

Rose

B, Keystone

JS, Taylor

Langer B. Diagnosis and management hydatid disease of the liver. Ann Surg 199:412-417.

Radiology

refrom

BR,

of 1984;

#{149} 145