J Gastrointest Surg (2014) 18:2057–2059 DOI 10.1007/s11605-014-2630-1

GI IMAGE

Hepatic Hydatid Cyst: A Rare Cause of Recurrent Pancreatitis William H. Kitchens & Charles Liu & Edward T. Ryan & Carlos Fernandez-del Castillo

Received: 1 August 2014 / Accepted: 7 August 2014 / Published online: 23 August 2014 # 2014 The Society for Surgery of the Alimentary Tract

Abstract A case of pancreatitis secondary to a hepatic hydatid cyst is illustrated together with its preoperative imaging and intraoperative appearance. Cystobiliary communication is a common complication of large hydatid cysts, and episodes of recurrent pancreatitis resulting from passage of cyst contents down the biliary tract are rarely described. The clinical manifestations, diagnostic workup, and surgical management options of echinococcal-related pancreatitis are discussed, and a review of the literature is provided. Keywords Pancreatitis . Hydatid cysts . Echinococcus

A 71-year-old man presented with a history of recurrent episodes of acute pancreatitis in the setting of a known large hepatic hydatid cyst. He had been raised in a rural area of the Middle East (immigrating to the United States 47 years ago), and his past medical history was notable for cutaneous anthrax and leishmaniasis as a child. Fifteen years ago, a ∼13 cm partially calcified hydatid cyst was incidentally noted in his right hepatic lobe, but no therapy was pursued. However, he subsequently developed episodes of acute pancreatitis requiring hospitalization in December 2000 and February 2002. Ultrasounds at the time found slight biliary sludge with no cholelithiasis. He endorsed minimal use of alcohol, and his serum triglycerides and calcium were both normal. He sought specialty consultation elsewhere for presumed gallstone W. H. Kitchens : C. Fernandez-del Castillo (*) Department of Surgery, Massachusetts General Hospital, ACC 366 15 Parkman St., Boston, MA 02114, USA e-mail: [email protected] W. H. Kitchens Emory Transplant Center, Emory University Hospital, Atlanta, GA, USA C. Liu Harvard Medical School, Boston, MA, USA E. T. Ryan Division of Infectious Diseases, Department of Medicine, Massachusetts General Hospital, Boston, MA, USA

pancreatitis, but he was advised against cholecystectomy due to the proximity of the gallbladder to his large hydatid cyst. He was subsequently asymptomatic until May 2013, when he again experienced severe epigastric and periumbilical pain, nausea, and vomiting. He sought evaluation at an outside hospital, and his initial labs were significant for an elevated lipase (2,008 units/L) and abnormal liver function tests (total bilirubin 4.5 mg/dL, alanine transaminase 657 IU/L, and aspartate transaminase 623 IU/L). He underwent a magnetic resonance cholangiopancreatography (MRCP), and it demonstrated no biliary ductal dilation or choledocholithiasis, but it did reveal development of a new 3.9 cm cyst lateral to his main cyst compared to prior images in 2005. It also suggested communication of the cyst with the biliary system (Fig. 1). He responded well to conservative management of his pancreatitis. Unexpectedly, while eosinophils comprised only 2.2 % of his white blood cells at the time of admission, follow-up labs obtained 10 days later revealed a marked eosinophilia (32.9 %). He presented to us 3 months after discharge. He had an entirely benign abdominal exam and no gastrointestinal complaints. However, as his marked eosinophilia could not be explained by simple gallstone pancreatitis, we entertained the possibility that his recurrent pancreatitis episodes could be due to cystobiliary communication with his hydatid cyst and subsequent pancreatitis from intermittent passage of cyst contents (“hydatid sand”) down his biliary tract. Although the option of endoscopic sphincterotomy was discussed with the patient, he opted for definitive surgical management given the formation of new daughter cysts on serial imaging.

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J Gastrointest Surg (2014) 18:2057–2059

Fig. 1 MRCP images of hydatid cyst, demonstrating a large (∼13 cm) multiseptated dominant cyst with cystobiliary communication

Fig. 2 Intraoperative findings after unroofing of hydatid cyst and evacuation of cyst contents (inset). Small area of active bile staining (white arrow) was seen in the cyst wall and oversewn

He was treated with 2 weeks of preoperative albendazole (400 mg PO bid) and 2 days of praziquantel (1,500 mg PO bid) before surgery. At the time of surgery, a right-sided subcostal incision was made and a large cystic structure (with two components) was identified on the inferolateral edge of the right hepatic lobe, adjacent to but not involving the gallbladder fossa. A cholecystectomy was performed without difficulty, and then the right hepatic lobe was mobilized by taking down the triangular ligaments and hepatic flexure. Having medialized the cysts, we isolated the operative field with laparotomy pads soaked in hypertonic saline as a scolicidal agent (as the patient had an allergy to povidoneiodine). We first attempted to aspirate the smaller, more calcified lateral cyst through a 12-mm trochar placed on the suction, but the cyst contents were very dense and paste-like. Part of the anterior cyst wall was excised and the cyst contents were carefully evacuated with a ring forcep. We then excised part of the anterior cyst wall of the adjacent larger cyst. The contents of this cyst were slightly bile-stained and less viscous, containing some intact gelatinous material consistent with scolices (Fig. 2, inset). We excised the shared wall between these two cysts, creating a common cavity to facilitate complete cyst evacuation. After thorough irrigation, we identified a single area of bile staining in the wall of the larger cyst, representing a cystobiliary communication (Fig. 2). This was oversewn with chromic suture, and no further bile leakage was encountered. The cyst cavity was packed with omentum and the abdomen was closed. The patient had an uneventful postoperative course and was discharged on postoperative day 6 to continue a 2-month course of albendazole. Final pathology of the cyst contents demonstrated abundant acellular laminated helminth cuticle with several armed scolices. Hydatid disease (cystic echinococcosis) is a parasitic disease most commonly caused by infection with the larva of Echinococcus granulosus, and it remains a formidable

clinical problem in cattle- and sheep-farming areas worldwide, in particular those in South America and in Mediterranean countries such as Turkey.1 The liver is chiefly involved in 70 % of cases, although many patients remain asymptomatic for decades due to the slow growth of the cysts. Eventually, many hepatic hydatid cysts become symptomatic due to mass effect, with hepatomegaly, right-upper quadrant/epigastric pain, and nausea being the primary symptoms.1 Many complications of hydatidosis result from fistulization of the cysts with the biliary tract. This cystobiliary communication occurs in 2–42 % of patients with liver hydatidosis and in up to 80 % of patients with cysts >7.5 cm in size.2 Although this cystobiliary communication is often occult, frank intrabiliary rupture of cysts can often provoke obstructive jaundice, cholangitis, and even anaphylaxis.2 Passage of hydatid cyst contents through the biliary tract is a rare cause of acute pancreatitis, and the limited case reports that exist in the literature almost all originate – in Turkey or the Mediterranean region.3 9 The proposed mechanism of pancreatitis is impaction of cystic debris (membranes and scolices) in the ampulla of Vater and pancreatic duct; alternatively, the pancreatitis may be an allergic response to echinococcal antigens in the papilla.6 Several groups have described endoscopic treatment of , , cystobiliary communication with sphincterotomy,2 10 11 although most published cases of echinococcus-related pancreatitis reported that patients ultimately underwent open surgical treatment with either liver resection or cyst fenestration and evacuation. This report is the first to simultaneously present the radiographic and intraoperative images of hydatid cyst-induced pancreatitis, an uncommon cause of recurrent pancreatitis that should be considered in patients living in echinococcus endemic areas.

J Gastrointest Surg (2014) 18:2057–2059

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2059 6. Chaudhary A, Upadhyaya AC, Kankanala VV, Kumar A, Joshi N, Kumar M, Harwani Y, Choudeswari RP. Intrabiliary rupture of hepatic hydatid cyst with impacted hydatid membranes at ampulla of Vater presenting as acute pancreatitis. Trop Gastroenterol 2013;34:43–44 7. Katsinelos P, Chatzimavroudis G, Fasoulas K, Kamperis E, Katsinelos T, Terzoudis S, Kokonis G, Patsis I. Acute pancreatitis caused by impaction of hydatid membranes in the papilla of Vater: a case report. Cases J 2009;2:7374 8. Sciumč C, Geraci G, Pisello F, Facella T, Vaglica R, Modica G. Acute pancreatitis during liver hydatidosis: treatment with ERCP and endoscopic sphincterotomy. Ann. Ital. Chir 2005;76:491–494 9. Wong LS, Braghirolli-Neto O, Xu M, Buckels JA, Mirza DF. Hydatid liver disease as a cause of recurrent pancreatitis. J R Coll Surg Edinb 1999;44:407–409 10. Khoshbaten M, Farhang S, Hajavi N. Endoscopic retrograde cholangiography for intrabiliary rupture of hydatid cyst. Dig Endosc 2009;21:277–279 11. Bedirli A, Sakrak O, Sozuer EM, Kerek M, Ince O. Surgical management of spontaneous intrabiliary rupture of hydatid liver cysts. Surg Today 2002;32:594–597

Hepatic hydatid cyst: a rare cause of recurrent pancreatitis.

A case of pancreatitis secondary to a hepatic hydatid cyst is illustrated together with its preoperative imaging and intraoperative appearance. Cystob...
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