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CASE REPORT
Year : 2022  |  Volume : 25  |  Issue : 1  |  Page : 114-117

A Case Report of Liver and Pancreatic Echinococcal Cysts


1 Department of General Surgery, Kartal Dr Lütfi Kırdar City Hospital, Istanbul, Turkey
2 Department of Surgical Oncology, Cukurova University, Balcali Hospital, Adana, Turkey

Date of Submission10-Aug-2020
Date of Acceptance11-Jun-2021
Date of Web Publication19-Jan-2022

Correspondence Address:
Dr. M O Gul
Department of Surgical Oncology, Cukurova University, Balcalı Training and Research Hospital, Adana
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/njcp.njcp_500_20

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   Abstract 


A 54-year-old female patient with complaints of abdominal pain for 2 months was admitted to the General Surgery clinic of our tertiary care hospital. Abdominal ultrasound (US) and computed tomography revealed cholelithiasis, liver hydatid cyst, and hypodense multicystic mass lesion in the pancreatic body. In the endoscopic US performed, pancreatic hydatid cysts were considered as the pre-diagnosis. Cystotomy and external drainage were performed on the 8-cm cystic lesion in the pancreas head-class junction. Pancreatic hydatid cyst can be rarely seen isolated or can develop synchronously to liver hydatid cyst, and should be kept in mind in a differential diagnosis. If the perioperative diagnosis is confirmed and in case of the absence of a pancreatic ductal fistula, surgical morbidity and mortality can be reduced by applying cystotomy and external drainage procedures.

Keywords: Echinococcus, hydatid cyst, pancreas


How to cite this article:
Uzunoglu H, Gul M O. A Case Report of Liver and Pancreatic Echinococcal Cysts. Niger J Clin Pract 2022;25:114-7

How to cite this URL:
Uzunoglu H, Gul M O. A Case Report of Liver and Pancreatic Echinococcal Cysts. Niger J Clin Pract [serial online] 2022 [cited 2022 Dec 3];25:114-7. Available from: https://www.njcponline.com/text.asp?2022/25/1/114/335985




   Introduction Top


Hydatid cyst caused by Echinococcus granulosis is an endemic public health problem in many parts of the world such as South America, the Middle East, Eastern Mediterranean, African countries, and China.[1] Although it can be seen in all body organs, the most common localizations are liver and lungs with an incidence of 70 and 12%, respectively.[2] Pancreatic echinococcosis is extremely rare and is seen in less than 1%.[3] Since the livers are the first filtering system for echinococcal embryos, most of the hydatid cysts settle in the liver. If the embryos are able to pass through the liver capillaries, they reach the lungs, the second filtering location. The embryos, which also succeed in crossing the pulmonary capillaries, can pass to the left side of the heart and reach all the body organs via the hematogenous way. Due to this natural course, the cyst can most commonly involve the liver, then the lungs, and then all the body organs such as the spleen, heart, muscle, brain, eyes, salivary glands, bone, urinary system, adrenal glands, breast, ovary, and pancreas.[4] Pancreatic echinococcal hydatid disease is a rare parasitic disease even in the areas where there is a high endemic disease. It can be seen with liver hydatid cyst or as isolated pancreatic echinococcosis. In the isolated primary pancreatic echinococcosis, the differential diagnosis may be difficult with other cystic lesions of the pancreas, or the diagnosis of hydatid cyst can be ignored.[2],[3],[4]

In this case report, we aimed to present the diagnosis and treatment approach of our case with primary pancreatic echinococcosis, which is a rare localization of hydatid cyst synchronously to liver cyst hydatid, and to review the literature information on this issue.


   Case Report Top


A 54-year-old female patient with complaints of abdominal pain and dyspepsia for 2 months applied to the General Surgery outpatient clinic. There was no feature in her history. On physical examination of the patient, epigastric tenderness was detected. Vital findings and other system examinations were normal. Laboratory findings were within the normal range. Among the tumor markers, the levels of carcinoembryonic antigen (CEA), CA19-9, and alpha-fetoprotein (AFP) were normal. Echinococcal Indirect Hemagglutination Test (Hydatidose, Fumouze Laboratoires, France) was positive. Abdominal ultrasound (US) revealed cholelithiasis, and a lesion compatible with type III Gharbi's Classification liver hydatid cyst with hyperechogenic appearance. In addition, a multicystic mass of 90 mm × 70 mm was observed in the section matching the pancreatic body localization, and it was considered to be a type III hydatid cyst. On computed tomography (CT), a cyst lesion of 110 mm × 88 mm was detected in segment-8 of the liver, and a hypodense cystic lesion of 60 mm × 50 mm was detected in the pancreatic body, and cholelithiasis, liver hydatid cyst, and pancreatic hydatid cysts were considered as the pre-diagnosis. However, since the pancreatic hydatid cyst is very rare, and the preoperative differential diagnosis of pancreatic cyst hydatid is difficult, endoscopic ultrasonography (EUS) was performed in the differential diagnosis, considering the cystic neoplasms of the pancreas, pseudocyst, and abscess. In the EUS, a cystic lesion with a double-wall structure, containing detached membranes and septations, was observed in the pancreatic class localization, and it was suspected to be a hydatid cyst [Figure 1]. The pancreatic parenchyma, duct, and choledoch were normal.
Figure 1: Double membrane image on endoscopic ultrasonography. In the endoscopic ultrasonography, a cystic lesion with a diameter of 58 × 60 mm, with detached membranes and septations, with a double-wall structure was observed in pancreatic body localization, and hydatid cyst was suspected

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The patient underwent laparotomy for diagnostic and therapeutic purposes. Cystotomy was performed on the 8-cm cystic lesion in the pancreas head-class junction. Upon detection of daughter vesicles and germinative membranes, the diagnosis of cyst hydatid was confirmed by frozen section examination. Since the diagnosis of cystic neoplasms was ruled out, radical resective procedures such as subtotal distal pancreatectomy are abandoned due to the location of the lesion close to the pancreatic head, and cystotomy and external drainage were performed [Figure 2]. Cholecystectomy was performed, and the operation was terminated by performing cystotomy and external drainage for the lesion in the liver dome. The patient was discharged with 10 mg/kg/day of albendazole treatment and control procedure. No recurrence was detected on CTs in the follow-up of the patient.
Figure 2: Hydatid cyst in the pancreatic neck. The patient underwent laparotomy for diagnostic and therapeutic purposes. A cystotomy was performed on a cystic lesion of approximately 8 cm in the head-trunk junction of the pancreas. Upon detection of the daughter vesicles and germinative membrane, the diagnosis of cyst hydatid was confirmed by frozen section examination. Cystotomy and external drainage were performed by abandoning radical resective procedures such as subtotal distal pancreatectomy since cystic neoplasms were excluded, and since the lesion was located close to the pancreatic head

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   Discussion Top


Hydatid cyst is an endemic public health problem that can be seen in many parts of the world and may involve each organ. Pancreatic echinococcosis is a very rare entity and is seen in less than 1%.[5] It can be seen as synchronous or isolated. Diagnosis of pancreatic cyst hydatid is usually made incidentally by CT and magnetic resonance imaging (MRI) scans.[6] About 50% of the cases are asymptomatic and diagnosed incidentally during radiological scans.[7] Pancreatic echinococcus may be presented with epigastric pain, abdominal mass, and with signs of acute and chronic pancreatitis, cholangitis, and obstructive jaundice and it may be asymptomatic. Its symptoms and complications depend on the diameter and the location of the cyst, and on the relationship between the cyst and the pancreatic duct.[5],[6],[7] In our case, there were no specific symptoms suggestive of pancreatic echinococcosis, except for epigastric pain. In our case, the hydatid cyst was detected in the liver, and a multicystic mass was detected in the pancreas body in the US and CT scans were performed for the epigastric pain etiology. At this point, pancreatic pseudocysts and pancreatic cystic neoplasms may be considered in the differential diagnosis, and there may be difficulty in the differentiation. In the literature, case reports in which pancreatic cystic neoplasia was considered in the radiological and the operative evaluation, but diagnosed as pancreatic echinococcus in the histopathological examination, were presented. Definitive diagnosis is made by surgery or histopathological examination in percutaneous drainage material. Differential diagnosis determines the treatment of choice, and is, therefore, important. In case of suspicion of malignancy, resective surgery should be preferred.[5],[6],[7],[8] In our case, a multicystic mass was described in the US and the CT evaluation, but cystic neoplasia could not be excluded in the differential diagnosis. In the EUS, a cystic lesion with a double-wall structure, detached membranes, and septations, with a diameter of approximately 58 mm × 60 mm was observed, and pancreatic echinococcosis was considered, and we got away from the diagnoses of other cystic lesions of the pancreas. Again, the fact that the pancreatic hydatid cyst was not isolated in our case and the presence of radiological characteristic cyst hydatid findings in the liver, which is the typical location for cyst hydatid, facilitated our preoperative differential diagnosis in favor of cyst hydatid.

US, CT, and MRI are frequently used in the diagnosis and evaluation of hydatid cysts. The most widely used test is the US due to its easy application and relatively cheap price. In societies where the disease is endemic, it is used for screening together with serological tests.[1],[2],[3],[8] CT and MRI can be useful in treatment planning by revealing the anatomical location of the cyst, the number of cysts, the presence of calcification, and female vesicles. In the evaluation of echinococcus, the sensitivity of the US for the liver is 90–95%, and the cysts are frequently observed as flat, round, and anechoic. Characteristic internal septations can be seen in the presence of female cysts. In the US, hydatid sand and cyst inner wall structure and double-membrane appearance are important. Abscess and neoplasms may be considered in the differential diagnosis in the absence of cyst membranes and mixed echo.[5],[8],[9] Difficulty of US evaluation of the pancreas and other imaging methods gain importance in cases such as obesity and gas distension. The sensitivity of CT is 95–100%, and it is more sensitive than the US in the diagnosis. CT is also superior to the US in terms of the detection of the number, size, and anatomical location of the cysts and the presence of an extrahepatic cyst. It is used in the follow-up of the lesions and in detecting the recurrence.[8],[10] MRI can define the cyst capsule better than CT. Except for infected and biliary-related cysts, it has no superiority over CT. MRI is also not cost-effective, and generally not required.[8],[11] CT and MRI are useful for lesions in extrahepatic locations such as the brain and pancreas.[12] Daughter cysts, hydatid sand, septation, and detached membranes are the characteristic radiological findings in the usual localization. In our case, EUS was very helpful in differential diagnosis, and besides the characteristic findings, the presence of a double membrane image in the cystic mass in the pancreas was evaluated in favor of a hydatid cyst.

Treatment methods of hydatid cyst include medical, percutaneous drainage, and surgical options. The goal of the treatment is to relieve pressure symptoms and pain, to prevent the development of infection, rupture, and anaphylaxis. Primary pancreatic hydatid cyst is a rare entity, and preoperative diagnosis is always difficult. Although medical and conservative methods are included in its treatment, the most appropriate and main treatment is surgery. Symptomatic and large cysts should be operated on before complications develop. It is generally appropriate to remove the entire cyst without organ resection.[13],[14] Surgery is reported as the main treatment in pancreatic echinococcus cases taking into account the location of the cyst and the presence or absence of a pancreatic ductal fistula. However, two large-scale systematic reviews revealed no consensus about the decision of radical or conservative surgery.[15],[16] Partial and total cystectomy, cystoenteric anastomosis, marsupialization, and external drainage procedures may be preferred. However, complete resection has been recommended to prevent a recurrence.[8],[13] The purpose of surgical treatment is the total removal of cyst contents. Pericystectomy is the ideal method. However, surgical morbidity and mortality should be considered in pancreatic hydatid cysts, and the continuity of postoperative endocrine and exocrine functions should also be ensured. While distal pancreatectomy is preferred in cases with corpus and tail locations, cyst excision and drainage procedures are preferred in cases with head and uncinate processes.[14] There are case reports in the literature where pancreatic body and tail cyst hydatid cysts mimicking cystic pancreatic neoplasm are treated with distal pancreatectomy and splenectomy.[8],[13],[14],[17] These case reports show that, although US and CT findings are characteristic hydatid cyst findings, distal subtotal pancreatectomy and splenectomy were performed in differential diagnosis due to isolation of the lesion and its location in an unusual location such as pancreas, and cyst hydatid was ignored in the differential diagnosis. In our case, cyst hydatid was considered in the foreground in the differential diagnosis since the lesion in the pancreas showed characteristic radiological cyst hydatid findings in the US, CT, and EUS, as they are not isolated but are synchronously compatible with cyst hydatid in the liver. In our case, cystotomy and external drainage were performed by avoiding more radical resections since (i) we got away from the diagnosis of cystic neoplasia of the pancreas, (ii) the diagnosis of hydatid cyst was confirmed with the detection of preoperative daughter vesicles, and (iii) the lesion was located close to the head of the pancreas. Thus, it was aimed to reduce morbidity and mortality, and to maintain the continuity of endocrine and exocrine functions of the pancreas.

As a result, pancreatic hydatid cyst can be rarely seen isolated or can develop synchronously to liver hydatid cyst, and should be kept in mind in the differential diagnosis. Benign and malignant cystic neoplasms of the pancreas, pancreatic pseudocyst, or abscess are included in the differential diagnosis. In addition to the US, EUS is also a very helpful imaging method in the preoperative diagnosis besides CT and MR. The presence of a double membrane image in EUS suggests the diagnosis of a hydatid cyst. The main treatment is radical or conservative surgery. When cystic neoplasms of the pancreas cannot be excluded, distal pancreatectomy can be performed in the pancreatic body and tail-located cases, and pancreaticoduodenectomy can be performed in the pancreatic head-located cases. If the preoperative diagnosis is confirmed and in case of the absence of a pancreatic ductal fistula, surgical morbidity and mortality can be reduced by applying cystotomy and external drainage procedures in the pancreatic head- and neck-located cases.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
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2.
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Ahmed Z, Chhabra S, Massey A, Vij V, Yadav R, Bugalia R, et al. Primary hydatid cyst of pancreas: Case report and review of literature. Int J Surg Case Rep 2016;27:74-7.  Back to cited text no. 3
    
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Geramizadeh B. Unusual locations of the hydatid cyst: A review from Iran. Iran J Med Sci 2013;38:2-14.  Back to cited text no. 4
    
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Kısaoğlu A, Özoğul B, Atamanalp SS, Pirimoğlu B, Aydınlı B, Korkut E. Incidental isolated pancreatic hydatid cyst. Turkiye Parazitol Derg 2015;39:75-7.  Back to cited text no. 5
    
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Bayat AM, Azhough R, Hashemzadeh S, Barband A, Yaghoubi AR, Gargari RM. Hydatid cyst of pancreas presented as a pancreatic pseudocyst. Am J Gastroenterol 2009;104:1324-6.  Back to cited text no. 6
    
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Kowalczyk M, Kurpiewski W, Zieliński E, Zadrożny D, Klepacki Ł, Juśkiewicz W. A rare case of the simultaneous location of Echinococcus multilocularis in the liver and the head of the pancreas: Case report analysis and review of literatüre. BMC Infect Dis 2019;19:661.  Back to cited text no. 8
    
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Wuestenberg J, Gruener B, Oeztuerk S, Mason RA, Haenle MM, Graeter T, et al. Diagnostics in cystic echinococcosis: Serology versus ultrasonography. Turk J Gastroenterol 2014;25:398-404.  Back to cited text no. 9
    
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El-Tahir MI, Omojola MF, Malatani T, al-Saigh AH, Ogunbiyi OA. Hydatid disease of the liver: Evaluation of ultrasound and computed tomography. Br J Radiol 1992;65:390-2.  Back to cited text no. 10
    
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Morris DL, Buckley J, Gregson R, Worthington BS. Magnetic resonance imaging in hydatid disease. Clin Radiol 1987;38:141-4.  Back to cited text no. 11
    
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Tüzün M, Altinörs N, Arda IS, Hekimoglu B. Cerebral hydatid disease CT and MR findings. Clin Imaging 2002;26:353-7.  Back to cited text no. 12
    
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Gündeş E, Küçükkartallar T, Çakır M, Aksoy F, Bal A, Kartal A. Primary intra-abdominal hydatid cyst cases with extra-hepatic localization. J Clin Exp Invest 2013;4:175-9.  Back to cited text no. 13
    
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Kütükçü E, Kapan S, Turhan AN, Ede B, Aygün E. Pancreatic hydatid cyst: Case report. Bakırköy Medi J 2005;1:74-6.  Back to cited text no. 14
    
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Dziri C, Dougaz W, Bouasker I. Surgery of the pancreatic cystic echinococcosis: Systematic review. Transl Gastroenterol Hepatol 2017;2:105.  Back to cited text no. 15
    
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Akbulut S, Yavuz R, Sogutcu N, Kaya B, Hatipoglu S, Senol A, et al. Hydatid cyst of the pancreas: Report of undiagnosed case of pancreatic hydatid cyst and brief literature review. World J Gastrointest Surg 2014;6:190-200.  Back to cited text no. 16
    
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Oruğ T, Akdoğan M, Atalay F, Sakaoğulları Z. Primary hydatid disease of pancreas mimicking cystic pancreatic neoplasm: Report of two cases. J Med Sci 2010;30:2057-60.  Back to cited text no. 17
    


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