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11 May 2024: Articles  Tunisia

Intrabiliary Rupture of a Hepatic Hydatid Cyst: A Case Report

Unusual clinical course, Challenging differential diagnosis, Diagnostic / therapeutic accidents, Management of emergency care, Unexpected drug reaction, Educational Purpose (only if useful for a systematic review or synthesis)

Maissa Jellali1ABCDEFG*, Hanen Zenati ORCID logo1ABCDEFG, Mohamed Zayati1ABCDE, Ibtissem Korbi1ADFG, Faouzi Noomen1FG

DOI: 10.12659/AJCR.943893

Am J Case Rep 2024; 25:e943893

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Abstract

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BACKGROUND: Hydatid disease is a common parasitic infection in many areas of Asia, South America, and Africa. It can affect any organ, most commonly the liver. The hydatid is often asymptomatic and the diagnosis is made when complications arise. The most common complication of this disease is opening in the bile ducts, which is a life-threatening condition causing serious acute cholangitis. We report a case of acute cholangitis caused by hydatid cyst rupture into the right bile duct.

CASE REPORT: A 33-year-old woman, with no medical or surgical history, presented to our Emergency Department with abdominal pain, jaundice, and fever for 3 days prior to admission. The patient was hemodynamically stable. In the examination, we noticed right upper-quadrant tenderness with guarding, icterus sclera, and negative Murphy sign. A CT scan showed a liver hydatid cyst of the 4th and 8th of segments, with intrahepatic and extrahepatic biliary duct dilation. The cyst communicated with the right hepatic bile duct via a large fistula. A diagnosis of acute cholangitis was made and she underwent conservative treatment with external drainage of the pericystic cavity through the biliary duct. The postoperative course was uncomplicated and she was discharged 15 days later.

CONCLUSIONS: The surgical approach to hepatic hydatid must be customized based on the specific characteristics of the cyst and associated complications. Acute hydatid cholangitis is a rare but serious complication of a hydatid cyst, which requires early diagnosis and adequate surgical management.

Keywords: Cholangitis, conservative treatment, Echinococcosis, Liver

Introduction

Hydatid disease (HD), also known as cystic echinococcosis (CE), poses a significant public health problem, particularly in regions with Tuzun livestock farming [1]. In endemic regions, CE has an annual incidence ranging from 1 to 200 cases per 100 000 individuals, with mortality rates that can reach 90% in untreated patients within 10–15 years after diagnosis [2]. HD is a parasitic infection caused by metacestode of the tape-worm Echinococcus granulosus. While the parasite can infect various organs [3], the liver is the primary site of involvement [4]. Hepatic hydatid cyst (HHC) is often asymptomatic and the diagnosis is usually made when complications occur. The most common complication of this disease is opening in the bile ducts [5,6]. Liver hydatid can rupture in any part of the biliary system. Small ruptures can remain occult or asymptomatic. However, large communication (greater than 5 mm) occurs in 3–17% of cases and causes severe cholangitis, which is a life-threatening condition [7]. The main treatment for HHC is surgery and the optimal surgical approach continues to be a subject of debate. The controversy surrounding radical versus conservative surgery for various conditions is a complex issue. This case report illustrates an acute cholangitis arising from a ruptured hydatid cyst in the liver. It underscores the potentially life-threatening nature of this complication and emphasizes the critical role of conservative surgery in achieving a successful outcome.

Case Report

A 33-year-old woman, with no medical or surgical history, presented to our Emergency Department with abdominal pain, jaundice, and fever beginning 3 days prior to admission. The patient’s background included employment in animal husbandry while living in a rural area with limited economic resources. The patient was hemodynamically stable. She was noted to have a temperature not exceeding 38°C (bacterial blood culture was not performed), blood pressure of 130/70 mm/GeneArt rate of 90/min, and respiratory rate of 20/min. In the examination, we noticed right upper-quadrant tenderness, with icterus sclera and negative Murphy sign.

The biological data showed an inflammatory syndrome with C-reactive protein at 180 mg/ml (reference value <8 mg/L), and white blood cells at 17000/mm3 (reference value 4–10×103/mm3). Furthermore, we noticed a biological cholestasis: hyperbilirubinemia with total bilirubin 65 umol/L (reference value <17 umol/L), direct bilirubin 48 umol/L (reference value <3 umol/L), and alkaline phosphatase was 261 U/l (reference value <45 U/L). The liver function test results were ALT: 375 U/L (reference value 5–45 U/L) and AST: 128 U/L (reference value 5–45 U/L). The other findings, including hemoglobin, urea, amylase, electrolytes, coagulation test, and albumin, were normal. Abdominal ultrasound was not performed and a CT scan was directly realized, showing a hydatic cyst of the 4th and 8th segments and a 14-mm common bile duct (Figure 1). This formation measured 84×76×100 mm, and it appeared to communicate with the right hepatic bile duct (Figure 2). A diagnosis of acute cholangitis was made, and we suspected that the biliary obstruction was directly caused by hydatid cyst rupture. The patient was hospitalized in the Surgery Department. She received medication for both the hydatid cyst and the cholangitis. The treatment regimen included albendazole at 10 mg/kg/day for the hydatid cyst and fluid resuscitation, ceftriaxone 1 g twice daily, and metronidazole 500 mg 3 times daily for the cholangitis.

The patient was operated on after 48 h with an under-right costal laparotomy by a senior surgeon in a university hospital. The perioperative finding was a hydatid cyst of segment IV and VIII and a dilated common bile duct. Following application of a scolicidal agent (hypertonic saline) in the operative area, an aspiration puncture of the cyst was performed, yielding a fluid with a bile-like appearance. Subsequently, a microbial bile culture was performed and an anterograde cholecystectomy was released. The perioperative cholangiography confirmed the large fistula between the right bile duct and the cyst (Figure 3). We performed a longitudinal choledocotomy, extraction of hydatid debris, and washing of the common bile duct with 0.9% NaCl solution (Figure 4). A choledocoraphy with a Kehr drain was released after verification of the vacuity of the VBP by a choledoscope. A hepatotomy was released and the hydatic material was extracted. The pericystotomy was securely sutured using slowly dissolving threads. Two Salem’s tubes were placed in contact with the hepatotomy. All laboratory findings returned to normal values during the first postoperative week and a microbial bile culture was negative. A postoperative cholangiography on day 10 and showed a fine common bile duct without lacunar image and a good duodenal passage. Therefore, the T tube drain was clamped at day 11, and the other drains were removed. The postoperative course was uncomplicated and the patient was discharged after 15 days. She received albendazole 400 mg twice daily for 3 cycles. Guided by our experience, the Kehr drain was removed 2 months after surgery after cholangiography (Figure 5), and CT scan (Figure 6) without complications. The patient was regularly monitored with an abdominal ultrasound and hydatid serology every 6 months. No recurrence occurred after 6 months.

Discussion

HD, originating from Echinococcus granulosus, is a zoonotic infection found mainly in the liver (50–70% of cases) [8]. The progression of Echinococcus granulosus in the liver is closely linked to the bile ducts. As the parasite grows, it can damage the integrity of the biliary system, causing biliary fistulas. In the case of a large fistula, debris from the ruptured cyst wall, along with cyst contents like daughter cysts (proliferating parasitic structures) and scolices (the infectious larval stage of the parasite), can block the bile ducts and cause acute cholangitis [9]. The large biliocystic fistula (LBCF) is defined by the presence of a fistula with a diameter greater than 5 mm. LBCF is called also frank intrabiliary ruptured [10]. LBCF causes severe obstruction of the biliary system and subsequent cholangitis. The LBCF complicates surgical management [11,12].

Clinical symptoms of LBCF are fever, abdominal pain, jaundice, pruritus, and pale stools [12]. Elevated liver function test results are expected when there is biliary contamination. The diagnosis of LBCF can be done with ultrasonography, which remains key for diagnostic examination. The ultrasound signs suggesting LBCF are dilatation of the intrahepatic bile ducts next to the liver cyst and presence of echogenic material in the extrahepatic duct without posterior acoustic shadowing [13]. CT has high sensitivity and specificity for hepatic hydatid disease, and MRI is also very helpful in diagnosis [13,14].

The treatment of this HD is always controversial. Endoscopic retrograde cholangiography (ERC) confirms the presence of a cysto-biliary communication and relieves the bile duct obstruction, but it cannot treat the cyst [15].

The curative treatment for HHC is surgery and the choice of surgical approach should be based on the presence or absence of a biliary fistula. Preoperative MRI can indicate the potential existence of a fistula. However, the definitive confirmation occurs during the intra-operative assessment, at which point the appropriate surgical procedure is chosen. Radical treatment is infrequently performed in endemic countries because of recurrence. Pericystectomy may be hazardous or even impossible to perform, especially for some localizations such as the dome or the paramedian sector. Conservative treatment is the criterion standard therapy for HHC [16].

The conservative treatment consists of evacuating the cyst, research of the biliary fistula by the review of hydatid liquid, whose bilious aspect shows a cysto-biliary communication [10]. The smoothing of pericyst crevices, followed by a perioperative cholangiography, can identify the location and diameter of the fistula. For LFBC, such as in our case, internal trans-Oddi’s sphincter fistula drainage is indicated, regardless of the location of the cyst and regardless of lateral or terminal type of fistula [17]. It involves drainage of the pericystic cavity through the biliary duct, either externally or internally within the digestive tube by suturing the pericystotomy. External pericysto-biliary drainage utilizes a Kehr tube, with its ascending branch passing through the biliary fistula to access the pericystic cavity [16,18]. Consequently, the remaining cavity efficiently drains into the bile duct through the biliocystic fistula.

Medical treatment (albendazole or mebendazole) is usually indicated before and after surgery to sterilize the fistulae and to avoid recurrences [19].

Conclusions

Rupture of a hydatid hepatic cyst into the biliary tree is a common complication of hydatid disease. Large fistulas caused by this rupture leads to acute cholangitis, which is a life-threatening condition. Accurate diagnosis and emergency surgical intervention are mandatory. Conservative surgery with cysto-biliary disconnection remains the method of choice in endemic areas and is a safe approach with favorable outcomes.

References:

1.. Aloqaily M, Al Hayek S, Shaf’ei M, A large splenic epidermoid cyst initially: Am J Case Rep, 2023; 24; e941585

2.. Wen H, Vuitton L, Tuxun T, Echinococcosis: Advances in the 21st century: Clin Microbiol Rev, 2019; 32(2); e00075-18

3.. Leitão P, Carvalho A, Fernandes T, Gonçalves J, Cystic echinococcosis: A case of extrahepatic intra-abdominal involvement: Case Rep Radiol, 2017; 2017; 8919546

4.. Xu Y, Hu X, Li J, Dong R, Hepatic hydatid cyst misdiagnosed as simple cyst: A case report: Am J Case Rep, 2020; 21; e923281

5.. Ramia JM, Figueras J, De la Plaza R, García-Parreño J, Cystobiliary communication in liver hydatidosis: Langenbeck’s ArchSurg, 2012; 397; 881-87

6.. Ghannouchi M, Rodayna H, Ben Khalifa M, Postoperative morbidity risk factors after conservative surgery of hydatic cyst of the liver: A retrospective study of 151 hydatic cysts of the liver: BMC Surgery, 2022; 22(1); 120

7.. Michalopoulos N, Laskou S, Papavramidis TS, Rupture of right hepatic duct into hydatid cyst: J Korean Med Sci, 2012; 27; 953-56

8.. Ito A, Nakao M, Lavikainen A, Hoberg E, Cystic echinococcosis: Future perspectives of molecular epidemiology: Acta Trop, 2017; 165; 3-9

9.. Elmajdoubi H, Elbarkaoui Z, Sebbah F, Rupture of hydatid cyst in the gall-bladder leading to acute cholangitis: Case Rep Infect Dis, 2021; 2021; 9858658

10.. Zaouche A, Haouet K, Jouini M, Management of liver hydatid cysts with a large biliocystic fistula: Multicenter retrospective study: World J Surg, 2001; 25(1); 28-39

11.. Fassi Fihri MJ, Lahkim M, Surgical treatment of complicated hepatic hydatic cyst: International Journal of Medical Science and Clinical Inventions, 2017; 4(5); 2953-56

12.. Pedrosa I, Saíz A, Arrazola J, Hydatid disease: Radiologic and pathologic features and complications: Radiographics, 2000; 20; 795-817

13.. Bektaş M, Dökmeci A, Cinar K, Endoscopic management of biliary parasitic diseases: Dig Dis Sci, 2010; 55; 1472-78

14.. Mehta P, Prakash M, Khandelwal N, Radiological manifestations of hydatid disease and its complications: Trop Parasitol, 2016; 6(2); 103-12

15.. Özaslan E, Bayraktar Y, Endoscopic therapy in the management of hepatobiliary hydatid disease: J Clin Gastroenterol, 2002; 35; 160-74

16.. Sozuer E, Akyuz M, Akbulut S, Open surgery for hepatic hydatid disease: Int Surg, 2014; 99(6); 764-69

17.. Toumi O, Ammar H, Gupta R, Management of liver hydatid cyst with cystobiliary communication and acute cholangitis: A 27-year experience: Eur J Trauma Emerg Surg, 2019; 45(6); 1115-19

18.. Sakhri J, Ben Ali A, Hydatid cyst of the liver: J Chir (Paris), 2004; 141; 381-89

19.. El-On J, Benzimidazole treatment of cystic echinococcosis: Acta Trop, 2003; 85(2); 243-52

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923