Hydatid disease is a worldwide zoonosis caused by the larval stage of the Echinococcus parasite. It represents a public health problem in endemic areas like in Tunisia [1]. Although hydatid cysts can
reach any organ of the human body, the liver is the most commonly affected organ [34]. Hydatid cysts of the liver are usually asymptomatic. However, some patients may become symptomatic due to complicated hydatid disease. In fact, Hydatid cysts of the liver may be complicated by infection, anaphylactic shock, compression of adjacent organs or rupture in the peritoneal cavity or biliary tract. Among all these complications, cysto-biliary communication is the most frequent one [35], leading, in some cases, to life-threatening cholangitis or cyst infection. However, acute pancreatitis is a little-known complication of hydatid disease. The primary pancreatic location of hydatid cyst actually represents less than 1% of all cases reported in the literature [36, 37]. It is responsible for a few cases of acute pancreatitis by cyst compression or migrated vesicle daughters’ obstruction of the main pancreatic duct [38]. Nevertheless, in our case, a cyst emerging from the liver has caused acute pancreatitis. In this case report, the pancreatic disease is related to the obstruction of the pancreatic duct by migrated parasite through the common bile duct. It implies accordingly the presence of a cysto-biliary communication. Almost all the well-documented reports of this rare complication are case reports.
Through a systematic Pubmed search, using the keywords “acute pancreatitis” and “liver hydatid cyst” and “hydatid pancreatitis”, we found 32 published articles dealing with the subject. We excluded 7 articles [2–7, 13] that could not be opened. We decided then to review 25 papers reporting 45 cases. The data has been analyzed using Excel. All the cases are summarized in Table 1. The main limitation of our analysis is that some cases lack important informations.
The mean age of the patients was 42 years with a range of 16 to81. The ratio of women to men was 0.48. Primary surgical treatment was performed in 11 patients, two of which were done in emergency cases. The treatment consisted in cystotomy or total pericystectomy without major liver resection. The common bile duct was explored and cysto-biliary communication was sutured in all cases. ERCP was a therapeutic alternative in 33 cases. Among these 33 cases, 12 patients hasn’t undergone surgery for the hydatid cysts whereas 21 had elective surgery. Emergency surgery was performed after endoscopic treatment in only one case. Nasocyst drainage was needed in one case and intrabiliary stent was put in 2 cases.
The symptoms weren’t specific and we couldn’t distinguish the hydatid cyst of the liver from other etiologies of acute pancreatitis at physical examination or patients’ blood tests. Serology could be suggestive of the hydatid origin of acute pancreatitis, but it is rarely made in emergency cases like in our patient. The diagnosis is hence set by either Ultrasonography or CT-scan. Ultrasonography is mainly made to clarify the etiology of acute pancreatitis. It is well-known that gallstones are the first etiology of pancreatitis. However, Ultrasonography is sufficient for the diagnosis of hydatid disease of the liver [39] but remains inadequate to assign it to the pancreatic disorder. In our case, Ultrasonography permitted us to suspect a hydatid cyst of the liver but we couldn’t confirm it’s liability in the pancreatitis etiopathogenesis. 5-days delayed CT scan is mandatory to identify the prognostic stage of acute pancreatitis and it was made in all the 45 cases reported in the literature. Furthermore, CT-scan could provide evidence of cysto-biliary communication or migrated daughter vesicles in the biliary tract, which is responsible for the occurrence of acute pancreatitis.
Open surgery for hydatid cysts ruptured in the biliary tracts remains the standard treatment. The general principles in treating hydatid cysts are applicable in the case of acute pancreatitis. The aim of surgery is to treat the cyst, the remnant cavity, the cystobiliary communication and to ensure the vacuity of the common bile duct by clearing the hydatid material. The procedure generally begins with the protection of the peritoneal cavity with scolicidal agents followed by the aspiration of the cyst content. We do not inject scolicidal agents in the cystic cavity in case of bilious content to prevent secondary biliary cirrhosis. Intraoperative cholangiography is performed to identify biliary fistula. Two methods [40] are commonly used to deal with hydatid cysts of the liver: A radical method consisting in total pericystectomy with the clearance of common bile duct and insertion of T-tube Kehr. A hepatectomy can also be performed but it is known to be a morbid procedure for such a benign disease. However, conservative surgery is the most commonly performed procedure and different techniques are described for the treatment of bliliary fistula according to their diameter. Cystotomy or partial pericystectomy and simple suturing of the fistula with drainage of the common bile duct using Kehr drain are reserved to fistulas less than 5 mm.On the other hand, for larger fistulas we can perform a trans-fistula oddien drainage (DITFO), biliary kysto-disconnection according to Perdromo, and bipolar drainage [35]. In our case, we decided to perform a bipolar drainage because of the efficiency and time-saving of this technique especially in case of emergency. Through the literature review, we found that 34 patients underwent open surgery, including emergency situations or delayed after ERCP and sphincterotomy, permitting the evacuation of membranes. Lahmidani et al. [33]reported 16 cases of hydatid acute pancreatitis.In this case series, all patients had ERCP permitting the evacuation of hydatid membranes in 81.25% of cases. All the patients underwent open surgery to treat definitively the cystic echinococcosis of the liver.
The main complication of liver hydatid disease surgery is biliary leakage that can lead to reoperation in some cases. The suppuration of the residual cavity is common and can be prevented by omentopolasty and capitonnage [40] .Anaphylactic shock is rare but described in few reports. However, postoperative course are usually uneventful like in our case.
ERCP with sphincterotomy is a promising diagnostic and therapeutic tool [14]. It permits to set the diagnosis of cystobiliary communication, evacuate hydatid material and eventually put a stent if the vacuity of the common bile duct is not assured. The endoscopic procedure is most commonly used in cases of obstructive jaundice or acute cholangitis. Furthermore, a postoperative ERCP is helpful for residual hydatid membranes left in the common bile duct. It is also useful for high anesthesia risk patients. In addition, endoscopic therapy can delay urgent and morbid surgery after sedation of the acute pancreatitis episode. However, Ghidirim et al. [20] reported a case of a 17-year old man who presented with an acute pancreatitis due to cysto-biliary communication of a hydatid cyst of the liver. He was treated with primary ERCP and extraction of hydatid membranes. Nevertheless, emergency surgery was mandatory two weeks later because of a new episode of hydatid rupture occurred with clinical presentation of cholangitis. This case report highlights the fact that radical or conservative surgical treatments of the cyst remain the only procedures aiming to eradicate the disease especially in low-income countries where ERCP is not affordable and hydatid disease is very common.