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Intrahepatic bile duct rupture of hydatid cyst: a severe complication for the patient
M. Stamatakos, K. Kontzoglou, S. Tsaknaki, C. Sargeti, R. Iannescu, C. Safioleas, M. Safioleas (Chirurgia, 102 (3): 257-262)
Introduction
Even since the times of Hippocrates people were aware of echinococcal disease as a life-threatening condition, caused by some kind of parasitic infection. Hippocrates himself described patients presenting "liver filled with water" (1-3). A tapeworm, occasionally infecting the human is responsible for this disease. There are four kinds of Echinococcus namely: E. Granulosus, E. Multilocularis, E. Vogeli, E. Oligarithrosis (4). The incidence of hydatid disease due to E. Granulosus is particularly high in sheep-raising countries such as Greece and the rest of the Mediterranean regions, Australia, South America, the far and the Middle East (5-10). In Greece about 8,1 in 100000 inhabitants suffer from the disease (11).

Incidence
E. granulosus is the most commonly responsible type for hydatid disease in Greece.
Infected fresh fruit and vegetables and dog's feces (definitive host) contain the parasite's ovi, and may be possible sources of infection for the human (intermediate host) (12).
The echinococcal tapeworm is naturally harbored inside the intestine of the definitive host where it is transferred from the gut of intermediate hosts, such as sheep, mouse, camels, horses, etc.
The parasitical eggs inside the human body, received by food, are directed towards the liver through the portal circulation.
The liver plays the role of a physical barrier which prevents expansion of the parasite to the rest of the body. However, in some cases the parasitical larvae continue their course towards the lungs and further through the arterial circulation. It may be installed in any organ, creating hydatid cysts.
Thus the liver is the most frequently impaired organ, followed by the lungs and, rarely, other human body tissues (13-24).
The disease may be manifested as unifocal or multifocal cysts, usually contralateral, most commonly localized on the anterior surface of the right lobe of the liver (11, 25).
In 30% of cases, hydatid disease presents one of the several possible complications, such as inflammation of the cyst or rupture within the peritoneal cavity, the biliary tree or other organs. Intrabiliary rupture of the hydatid cyst is the most frequently seen complication during disease progression (26), described as well by the terms "cystobiliary fistula" and "cystobiliary communication". Surgical management is the only solution for hydatid disease, using a variety of operational methods. However, the choice of a "gold standard" method remains disputable.

Clinical image
The size and number of the cyst, its localization and additional complications are the main factors determining the specific clinical manifestation.
Hydatid cysts of the liver generally enlarge by a mean of 1-5 cm per year, and become symptomatic when their diameter exceeds 5 cm (24); even cysts as large as 50 cm have been described.
The classic case concerns a small sized cyst asymptomatic for several years which is accidentally revealed during radiography or ultrasonography control conducted for an irrelevant reason.
In case of symptoms, these consist of a mild dull pain at the right upper quadrant, as well as abdominal discomfort.
Abdominal physical examination usually reveals hepato-megaly, a palpable mass in the liver region, while percussion may give the specific sense of the hydatid cyst (27).
If the cyst is located at the diaphragmatic region, pleural pain accompanied by cough may be present (28).
Rarely the disease concerns the inferior and posterior surface of the liver, adjacent to the major hepatic veins entering the inferior vena cava. This form of hydatidosis may cause pressure or incomplete obstruction of these major vessels, leading to associated symptomatology (29, 30).
During the course of echinococcosis three major complications may appear: purulence, rupture or calcification of the hydatid cyst. The patient presents an acute aggravation of his clinical condition with clear signs and symptoms, which contribute to the differential diagnosis.
Purulence of the hydatid cyst is usually due to micro-organisms parasitically developed in the blood, which colonize the cyst via a hematogenous infection thus causing the pathogenic factors. Moreover, a cyst may be infected after minimal ruptures creating a communication with adjacent small bile ducts. This complication is clinically manifested by high fever, hepatic pain, acute weight loss and jaundice. Differential diagnosis should exclude the case of hepatic abscess formation, an entity of a more obscure prognosis (31).
Various types of rupture of a hydatid cyst are possible, the most severe one being rupture towards the bile ducts, observed in 12-18% of patients presenting echinococcosis (32). Potential causative factors are increased pressure inside the cyst, as already mentioned, inflammation, minimal hepatic injury, or a significantly elevated hydrostatic tension of the cyst compared to the respective bile duct tension (33-35).
The pathophysiologic base of this menacing complication is related to pressure exerted by the hydatid cyst to the surrounding parenchyma usually in case of centrally located cysts, to cystic content escaping to some smaller or larger bile duct. Rupture may include the common hepatic duct, lobar bile branches, or small intrahepatic bile ducts. An additional provocative factor contributing to cystic wall rupture is an endocystic pressure of more than 80 cm H2O (36).
A hydatid cyst complicated by rupture is mainly manifested by obstructive jaundice, fever associated with rigor, biliary type of pain, abdominal discomfort and allergic reactions (32, 37, 38).
Three main clinical forms of this complication are identified: contained, direct and communicating rupture (39). Rupture of the endocyst alone with limitation of cystic fluid behind the pericystic wall represents the first type. In case of a direct rupture, the larvae are spilled inside the peritoneal cavity, leading to chemical peritonitis. Rarely, the hepatic capsula prevents spillage of cystic content, providing a physical barrier in cases of subcapsular rupture. Moreover, given a previous rupture of the endocyst, during the expansion of the hydatid cyst, its wall is frequently incorporated by the surrounding tissue, resulting in diffusion of the cystic fluid into adjacent bile ducts. These effects constitute the communicating type of rupture, observed in the majority of cases. The communication between the cyst and bile ducts is created either by small incisions of the cystic wall or fistula formation, or it may be due to simple rupture inside a main branch of the biliary tree (40). As far as the common bile duct is concerned, a diameter of 5 cm is considered the baseline above which intrusion of the cystic content inside the biliary tree is rendered possible, leading to related symptoms (41). Considering the higher rigidity of greater bile duct wall, communicating rupture and diffusion of the hydatid fluid is more likely to occur inside bile ducts of a smaller diameter. Implication of greater bile ducts is manifested by obstructive jaundice, and less frequently by acute cholangitis due to impairment of the whole biliary tree. Ultrasonography control provides rapid and accurate diagnosis, while ERCP contributes to identification and treatment of the whole entity by imaging and endoscopic sphincterotomy. Following confirmation of the diagnosis, early management is essential in order to prevent the deleterious consequences of this complication.
Among a smaller group of patients the hydatid cyst is ruptured towards the pleura and the bronchial tree, due to pressure created by continuous cyst enlargement. In this case patients present pleuritis purulent with or without anaphylactic reactions. In addition, intense pleural pain of the right basal region, paroxysmal coughing, bronchospasm, an intense feeling of choking, cyanosis, hydatidemesis and fever constitute possible symptoms.
The appearance of sputum containing bile is also indicative of cystic communication with bile ducts (32, 33).
Free rupture of a hydatid cyst and spillage of its content into the peritoneal cavity may follow abdominal injury. This is a rather rare effect during the progression of the disease, usually concerning cysts located on the inferior surface of the liver. Intraperitoneal dissemination of the hydatid debris is followed by the creation of a maternal cyst located in the small pelvis, or the appearance of multiple smaller cysts all over the peritoneal cavity. Clinical manifestations include diffuse abdominal pain, peritonitis, choloperitonitis and anaphylactic shock (34).
Calcification of the hydatid cyst is a rather benign evolution of the disease, included among the complications of echinococcosis.

Diagnosis
Preoperative control does not always ensure definitive diagnosis of the disease, since in several cases the diagnosis is possible only during surgery, or postoperatively through histological examination. Co-estimation of a detailed history, a careful clinical examination and a fully conducted laboratory assessment are essential. A preoperative differential diagnosis from obstructive jaundice due to cholelithiasis is in the majority of cases extremely difficult and the diagnosis is established intra-operatively. The use of various imaging techniques, such as US, CT, MRI, are favored in order to demonstrate the hydatid cyst, with ultrasonography and computed tomography being considered as the most sensitive ones. Ultrasonography is the diagnostic method of choice to reveal the cystic nature of the mass, imaging an echogenic lesion, although it does not speficy the type of rupture (42). Suspicion upon penetration of the hydatid cyst is based on the accurate imaging of the communication, which is very difficult to be revealed using the usual imaging techniques (43, 44), and is evidenced in only 25% of cases with absence of obstructive jaundice. Simple radiography may demonstrate the presence of the hydatid cyst inside the gallbladder or the common bile duct (45-47). ERCP or percutaneous transhepatic cholangiography (PTC) are more sensitive methods for revealing rupture of the cyst towards the hepatic bile ducts, although PTC has demonstrated a high risk of anaphylactic reaction. Moreover ERCP can not be conducted in every patient, and is applied under specific indications such as preoperative acute cholangitis due to presence of daughter cysts or necrotic hydatid tissue.
Multiple laboratory and serological tests have proven to be helpful and contribute to the detection of the disease and the determination of its stage. They may demonstrate eosinophilia, present in 50% of cases associated with rupture, positive echinococcal antibodies, positive Casoni or Weinberg tests, while ELISA immunoassay and test of indirect agglutinin can be conducted as well (48-53). None of these tests however provide a reliable predictive factor of the possibility of cystic rupture inside the bile ducts. Although several studies demonstrated a positive Casoni test in 88% of cases, while 85% positive results of ELISA have been mentioned, it is certain that these examinations present a high range of false negative results (20%), which the reason why their use is no longer applied.
When rupture inside the biliary tree occurs, severe consequences for the patient will follow. Thus in this case primitive diagnosis and management is vital, in order to achieve an improved outcome of these patients' health.

Treatment
Conservative treatment, surgical management or combination of methods, all have been suggested as adequate approaches to the hydatid disease.
If surgery is not a possibility, conservative management with administration of antihelminthic agents per os, such as albendazole, mebendazole or praziquantel may provide effective results (54-59). Another indication for the use of the above mentioned drugs is during the preoperative course in order to minimize the toxicity of the parasite and thus reduce the possibility of peritoneal infection during surgical dissection of the cyst, followed by dissemination of its content in the peritoneal cavity. Moreover, antihelminthic agents are proved useful during the postoperative period because they decrease the risk of recurrence.
Surgery is the method of choice, providing a unique possibility of definitive cure, thus applied for the management of any resectable hydatid cyst, despite its specific localization on the liver.
In order to achieve effective management of a hydatid cyst complicated by intrabiliary rupture, management of the cyst alone or biliary clearance limited in the common bile duct, are certainly inadequate. The operation is usually conducted through a right sub-costal incision. Classic surgical procedure includes drainage of the cystic content, management of the cavity, and evacuation of hydatid fluid from the biliary tree and, finally, restoration of normal bile flow. Initial isolation of the cyst from the peritoneal cavity is essential to prevent intra-peritoneal spillage of cystic content. After evacuation of the cystic cavity, the use of scolicidal agents is indicated, in order to minimize the possibility of complications due to peritoneal contamination, prevent recurrence of the disease, thus improving the patient's postoperative course. Solutions such as hypertonic saline 13-30%, hydrogen hyperoxide, alcohol and silver nitrate solution have been used for this purpose (60). This way, complications such as biliary leakage, hemorrhage or abscess formation are unlikely to occur.
A variety of methods have been suggested for the management of the residual cavity, the most effective ones being omentoplasty or marsupialization.
In cases of inflammatory cysts, external drainage methods should be applied, although they may be responsible for various complications.
Several radical approaches have also been applied, presenting a successful outcome, such as selective hepatic lobectomy, total cystectomy or partial cystectomy followed by Roux-en-Y pericysto-jejunostomy.
Hepatic lobectomy is indicated in cysts affecting one lobe only or in case of multiple large cysts. Partial hepatic resections are generally performed when destruction of a whole lobe caused by enlargement of a single or multiple cysts associated with reactive hypertrophy or the opposite lobe, are diagnosed. These procedures limit postoperative complications and possibility of residual disease.
A less radical, although not a conservative method, is total cystectomy, effective for hydatid cysts of a small or medium diameter. This type of management allows preservation of the hepatic parenchyma, combined with minimization of the recurrence rate. Partial cystectomy with internal drainage through a Roux-en-Y pericysto-jejunos-tomy constitutes and alternative for large cysts in patients with a severe general condition.
Primary management of the hydatid cyst is followed during the operative procedure by exploration and drainage of the common bile duct. Clearance of hydatid debris inserted into the biliary tree because of rupture is vital to prevent development of acute cholangitis or septic peritonitis and shock (61, 62). There is an ambiguity concerning the preferable procedure for this purpose in case of pre or intra-operative diagnosis of the complication. The treatment applied depends on whether cystic elements inside adjacent bile ducts are detected. In case of a visible incontinence of the cystic wall, application of sutures on the incision is recommended if cystic content is not present inside normally sized bile ducts. When leakage is observed, choledochotomy combined with evacuation of the biliary tree from cystic elements with or without the use of 0,9% NACL solution and T-tube drainage are strongly suggeested. Regarding T-tube insertion, this is a method favored by several authors even in patients with normal Oddi sphincter and common bile duct function. Its positive effects include direct control of biliary drainage during the postoperative period, thus improving the follow-up of a disease with poor prognosis. T-tube rather than cholopeptic anastomosis or sphincteroplasty is preferred due to a smaller perioperative mortality rate. When cystic debris is detected inside the common bile duct or the gallbladder, choledocho-duodenostomy is the only choice for radical treatment, in order to cover the possibility of additional hydatid cysts formation inside the common bile duct. In this case choledochotomy only would be an inadequate approach. Some authors suggest that patients with good general condition could successively undergo sphinctero-tomy or sphincteroplasty (63).
Patients presenting additional cholelithiasis or cholecystitis, as well as with hydatid cyst located next to the gallbladder, are the indications for additional cholecystectomy (62), otherwise the impaired gallbladder remains a continuous risk of future acute cholecystitis, even if no gallstones are present.
Surgeons should not forget the additional therapeutic role of endoscopic diagnostic techniques, easily and effectively applied in hydatid disease. Indications for the use of ERCP are cholangitis due to cystic material, incomplete evacuation of the biliary tree from cystic elements and prolonged biliary fistula caused by papillary stenosis (64, 65). Endoscopic sphincterotomy (EST) may prove useful in the management of persistent external biliary fistulas.
In case of biliary-cystic fistulas that were not managed during operation, transphincteric evacuation of the common bile duct combined with sphincteroplasty is an effective possibility.
It should finally be mentioned that common mortality causes among these patients are sepsis and hepatic failure (66, 67).

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