H Ono, M Okabe, T Kimura, M Kawakami, K Nakamura, Y Danjo, K Nagashima
H Ono, M Okabe, T Kimura, M Kawakami, K Nakamura, Y Danjo, K Nagashima. Role of Therapeutic Endoscopy in Hepatic Hydatid Disease after Surgical Intervention: Case Report. The Internet Journal of Gastroenterology. 2010 Volume 10 Number 2.
A 79-year-old female diagnosed as hepatic hydatid disease after hepatectomy was referred with jaundice and right hypochondralgia. An endoscopic retrograde cholangiography (ERC) showed a defect in the bile duct. We performed bile duct stenting, but stent occlusion occurred repeatedly. Bile duct stenting was performed 7 times and endoscopic naso-biliary drainage once. Although bile duct stenting was performed repeatedly, ERC provided an excellent diagnostic and therapeutic modality in the present case; thus, it should be considered as a definitive treatment in similar cases, particularly after hepatectomy or if the surgical risk is anticipated to be high.
Hydatidosis is a zoonosis that is generally caused by infection with
A 79-year-old woman with jaundice and right hypochondralgia was referred to our
hospital in January 2008. Right hepatectomy was performed in April 2003 because of
hepatic hydatid disease, and the margin was found to be positive for hydatid cysts. Her
height and weight were 151 cm and 42 kg, respectively; she had been treated with
Physical examination revealed right hypochondralgia and jaundice; her body
temperature was 37.6 ℃. Her white blood cell count was 12,900 /μL, and the levels of
C-reactive protein, alkaline phosphatase, and total bilirubin were 12.4 mg/dL, 1,135
U/L, and 18.5 mg/dL, respectively. Abdominal ultrasonography showed a dilated
intrahepatic bile duct, and abdominal computed tomography (CT) showed lesions with
calcifications and a dilated left intrahepatic bile duct (Fig. 1). Magnetic resonance
cholangiopancreatography revealed the obstruction of a 2 cm long bile duct (Fig. 2). A
diagnosis of acute cholangitis with obstructive jaundice was made, and intrabiliary
rupture of a relapsing hydatid cyst was suspected to be the cause of this condition.
ERC (Fig. 3A, B) and bile duct stenting (Cotton-HuibregtseⓇ Biliary Stent Sets;
Wilson-Cook Medical, Inc., NC, USA) were performed on January 22, 2008 (Fig.
4A-D), and the intracystic fluid and debris drained to the bile duct stent. Two months
after bile duct stenting, the level of total bilirubin was 1.5 mg/dL, and no dilated
intrahepatic ducts were observed on abdominal CT scans (Fig. 5). The patient was
discharged in March 2008; however, she was referred to our hospital again in July 2008
with jaundice and right hypochondralgia probably because of bile duct stent occlusion.
The patient was admitted to our hospital 5 times during a period of 3 years, and bile
duct stenting was performed 7 times, while endoscopic naso-biliary drainage was
performed once. The last bile duct stenting (plastic stent; 8.5Fr, 12 cm) was performed
in September 2010, and she survived for a period of 7 years and 8 months after right
Intrabiliary rupture of a hepatic hydatid cyst is a common complication and may
occur in 2 forms: an occult rupture, in which only the cystic fluid drains to the biliary
tree and is observed in 10-37% of the patients; and frank rupture, which has an overt
passage of intracystic material to the biliary tract and is observed in 3-17% of the
patients (3, 4). Intrabiliary rupture mainly occurs in centrally localized cysts, and an
intracystic water pressure up to 80 cm is also a predisposing factor for the rupture (5).
Intrabiliary rupture occurs in the right hepatic duct (55-60% cases), left hepatic duct
(25-30% cases), hepatic duct junction, common bile duct (CBD), or cystic duct
(8-11%); perforation into the gallbladder may be observed in 5-6% of cases (6-8). It was
thought that the present case was an occult rupture, because the intracystic fluid and
debris drained to the bile duct stent.
Intrabiliary ruptures of hydatid cysts have been diagnosed by imaging and
laboratory tests. Although these tests were proven to be ineffective in detecting
occult ruptures, certain findings from studies conducted in the USA provide essential
clues for the diagnosis of frank ruptures with obstruction. Echogenic material, without
posterior acoustic shadowing in extrabiliary ducts, implied the presence of intracystic
material (9). An abdominal CT scan may reveal a dilated CBD with low attenuation
intraluminal material, suggesting the presence of hydatid sand and cysts (10, 11).
Some cases of definitive endoscopic treatment of obstructive jaundice after
intrabiliary rupture have been reported; ERC has become the “gold standard” for the
assessment of intrabiliary rupture by achieving a detection rate of 86.6% to 100% (3,
12-15). ERC, which is a minimally invasive procedure, may obviate reoperation.
Endoscopy is a modality serving both diagnostic and therapeutic aims. Postoperative
ERC has several advantages: I-clarification of the causes of ongoing or recurrent
symptoms and laboratory abnormalities; II-resolution of obstructions or cholangitis due
to residual material in biliary ducts; III-management of postoperative external biliary
fistulae; and IV-provision of a realistic solution for secondary biliary strictures (12, 13,
ERC with bile duct stenting is performed for the treatment of intrabiliary ruptures
associated with obstructive jaundice, but this is rare in Japan. Ichushi Web, a Japanese
medical database, showed that only 4 Japanese cases, including the present case, were
reported from 1983 to 2010 (Table 1). In all cases, bile duct stenting was repeatedly
performed because of stent occlusion, but major complications were not encountered
after endoscopic procedures.
In conclusion, therapeutic endoscopy is a safe and valuable procedure for the
postoperative management of patients with hepatic hydatid disease.