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 Table of Contents  
DISPATCHES
Year : 2021  |  Volume : 11  |  Issue : 1  |  Page : 42-45  

Fasciola hepatica association with gallbladder malignancy: A rare case report


1 Department of Microbiology, SLN Medical College & Hospital, Koraput, Odisha, India
2 Department of Microbiology, Institute of Medical Sciences and SUM Hospital, S ‘O’ A Deemed to be University, Bhubaneswar, Odisha, India
3 Department of Gastroenterology, Institute of Medical Sciences and SUM Hospital, S ‘O’ A Deemed to be University, Bhubaneswar, Odisha, India

Date of Submission17-Mar-2020
Date of Decision23-May-2020
Date of Acceptance30-May-2020
Date of Web Publication13-May-2021

Correspondence Address:
Sarita Otta
1/16, Kanchanjunga Enclaves, Chandrasekharpur, Bhubaneswar, Odisha
India
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DOI: 10.4103/tp.TP_25_20

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   Abstract 


Fasciolopsis is one of the rarest parasitic infestations in our locality. The usual definitive host is the sheep; humans are accidental hosts in the life cycle of a liver fluke – Fasciola. In the chronic phage of Fasciolopsis, the patient presents with cholestasis and cholangitis. Yet, there is no proof of association of this parasite with carcinoma of the gallbladder. We here present such a case of Fasciolopsis in association with Stage IV gallbladder malignancy. Fasciola worms were extracted on endoscopic retrograde cholangiopancreatography done as a palliative measure for associated obstructive jaundice. The chronic phase of this zoonotic infection can be easily misdiagnosed as any other cause of obstructive jaundice if not properly investigated. The importance of repeated stool examination for parasitic ova and cysts should never be understated as it may help in early diagnosis of such treatable conditions as well as preventing the complications.

Keywords: Endoscopic retrograde cholangiopancreatography, Fasciola hepatica, gallbladder malignancy


How to cite this article:
Swain B, Otta S, Sahu MK, Uthansingh K. Fasciola hepatica association with gallbladder malignancy: A rare case report. Trop Parasitol 2021;11:42-5

How to cite this URL:
Swain B, Otta S, Sahu MK, Uthansingh K. Fasciola hepatica association with gallbladder malignancy: A rare case report. Trop Parasitol [serial online] 2021 [cited 2021 Jun 25];11:42-5. Available from: https://www.tropicalparasitology.org/text.asp?2021/11/1/42/315928




   Introduction Top


Human fascioliasis, a trematodal zoonotic infestation, rarely reported from this part of India. Humans become incidental hosts for liver flukes acquired by ingesting contaminated aquatic vegetations. Many cases are asymptomatic, thus have a chance of being missed unless thorough examination is not carried out. Here we describe a case of Fasciolosis associated with inoperable Carcinoma Gall Bladder.


   Case Report Top


A 45-year-old female presented to IMS and SUM Hospital, Bhubaneswar, with gradually progressing symptoms of yellowish discoloration of sclera, reduced appetite, pain abdomen with vomiting, and weight loss for 2 months. On examination, she was conscious and oriented with marked icterus and no pallor. The abdomen was soft on palpation and elucidated tenderness, particularly in the right hypochondrium. The liver function tests were deranged with conjugated hyperbilirubinemia, bilirubin in urine, and raised prothrombin time. She was a rural resident, illiterate, nonobese, nondiabetic person. She had no family history of any malignancy. She did not have any history of smoking and alcoholism and no history of enteric fever in the recent past. Her bile culture was sterile, and she was seronegative for HIV, hepatitis C virus, and hepatitis B virus. Her routine single stool examination was negative for any parasite. Ultrasonography (USG) of the abdomen showed a lobulated outline of gallbladder (GB) neck and body and growth with contiguous hepatic infiltration forming a mass. There was infiltration of primary biliary confluence with moderate bilobar intrahepatic biliary radical (IHBR) dilatation and infiltrated second part of the duodenum as well as multiple metastatic lesions in the liver. Contrast computed tomography (CT) showed hepatosplenomegaly with IHBR dilatation with abrupt tapering in the middle part of the common bile duct (CBD). The gallbladder was over distended with three irregular soft-tissue densities around 4.1 cm × 2.6 cm in the lumen. Thus, a preliminary diagnosis of inoperable Stage IV carcinoma gallbladder metastasizing to the liver was put forth. Endoscopic retrograde cholangiopancreatography (ERCP) was planned for biliary cannulation with stenting as a palliative measure. Astonishingly, multiple adult worms were visualized in the duodenum on ERCP [Figure 1], two of which could be recovered during the procedure while the other two escaped into the large intestine. The isolated parasites were large approximating 25–30 mm × 12–15 mm, flat, brownish, and leaf like [Figure 2]. Each worm's posterior end was pointed, and the oral sucker was situated in the conical projection at the extreme anterior end of the organism. Thus, it was morphologically identified as Fasciola hepatica. We did not find any eggs in her stool or those of the closely associated family members on careful direct examination and by concentration technique.
Figure 1: Flatworms found during endoscopic retrograde cholangiopancreatography

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Figure 2: Recovered adult worms of Fasciola hepatica during biliary stenting

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Since the patient had an inoperable tumor, she received palliative therapy for advanced stage of gallbladder carcinoma. She received antihelminthic drug albendazole for parasite clearance following which she was discharged on request.


   Discussion Top


Fascioliasis, the liver fluke disease of sheep, cattle, and other herbivorous animals, is virtually seen throughout the world. Humans are accidental hosts for the causative parasite, F. hepatica.[1] Infection results from ingestion of metacercariae present on uncooked and unwashed vegetables (e.g., watercress and sorrel).[2] In our case, the patient was illiterate and may have had the habit of using improperly cooked and unwashed vegetables leading to the disease.

Human Fasciolopsis in India is extremely uncommon, with only a few cases reported so far from North, North Eastern India, Mumbai, and Vellore. Animal studies indicate that Fasciola gigantica is more prevalent in India than F. hepatica.[2] Both the worms can be differentiated based on the morphological analysis. F. hepatica is a large, flat, brownish worm, measuring 30 mm × 13 mm.[3],[4] The size of F. gigantica is more than double (about 5 cm) that of F. hepatica, and the posterior end of the F. hepatica is pointed, whereas F. gigantica is rounded.[5] Although morphologically we identified the parasite as F. hepatica, further confirmation of species can be done by molecular studies.

After ingestion of aquatic vegetables contaminated with larvae of Fasciola hepatica, they excyst and pass through the intestinal wall into th peritoneum. They penetrate the Glisson's capsule and find their way through the liver to the bile ducts, where they reside as adult worms. Typical symptoms can be divided by phase of the disease: acute or liver phase and chronic or biliary phase. The first acute or liver phase lasts from 3 to 5 months and is caused by the migration of immature larvae from the duodenum to the liver. Symptoms include prolonged fever, hepatomegaly causing abdominal pain, and mild eosinophilia. Other manifestations are anorexia, weight loss, nausea, vomiting, cough, diarrhea, urticaria, lymphadenopathies, and arthralgia.[4] The chronic or biliary phase begins after approximately 6 months when the parasite matures in the bile ducts. It may last several years (>10 years) and is asymptomatic in more than half of the cases. When symptoms appear, these reflect commonly biliary obstruction with upper abdominal pain, intermittent jaundice, and extrahepatic cholestasis.[6],[7] In approximately 50% of infected humans, the presentation can be subclinical.[7] The patient's acute signs and symptoms may have gone unreported. We received the patient in the stage of extrahepatic obstruction and carcinoma.

While chronic inflammation is connected to increase cancer rates, it is unclear whether Fasciolopsis is associated with increased cancer.[8] In one of the meta-analyses,[8] only five in vitro animal studies have shown the association of Fasciola and cancer, while none of the human cases of cancer were explained by Fasciola. Animal studies show that the parasite is able to induce DNA damage through the action of mutational mediators such as reactive nitrogen species and reactive oxygen species.[9] In preliminary reports, F. hepatica has been shown to cause hepatic damage by inducing fibrosis in the liver parenchyma and by upregulating the genes for liver fibrosis and cirrhosis in infected animals,[10] and liver fibrosis is a widely known major risk factor for the development of liver cancer.

Factors predisposing to gallbladder malignancy are American nativity, family history for cholangiocarcinoma, gallbladder stone or polyp, age above 70 years, obesity, female sex, and diabetes mellitus.[11] Apart from female gender, we did not find any other risk factor in our patient. Rather, we detected multiple number of F. hepatica in the biliary tract. Therefore, we propose that the parasites may have led to chronic gallbladder irritation leading to the malignant changes.

Diagnosis is based on microscopic identification of the characteristic eggs in the feces or bile. In humans, maturation and excretion of the eggs take 3–4 months.[12] They are large, oval, yellowish-brown, operculated ova.[2] Demonstrating the eggs in stool sample may need repeated examination with concentration procedures. We did not find any eggs in her stool or those of the closely associated family members. Repeated stool examination was not possible as the patient was too sick. Nonspecific signs such as eosinophilia and raised erythrocyte sedimentation rate were not present in our patient. Her important signs were those of deranged liver function tests and gradually deteriorating kidney function.

One of the reasons for F. hepatica being reported less than its actual prevalence in India could be due to the nonavailability of CT imaging as well as inaccurate interpretation of the CT findings.[13] USG, which is the usual investigation, may show nonspecific finding as hypoechoic areas in the liver which is generally found in hepatic neoplasia.[14] Our patient also had indeterminate findings on USG. CT scan shows characteristic grape-like clusters of cysts “Fasciola cluster of grapes sign,” representing granulomas in the biliary distribution radiating from the liver capsule where the parasite enters the central biliary tree. Oblique reformations are especially useful for the demonstration of the above findings. Only F. hepatica can produce the “Fasciola tunnel sign” which is a specific sign on CT making it a definitive imaging tool for F. hepatica. A contrast-enhanced MRI may show similar findings but is less easily available and less amenable to dynamic multiplanar reformations.[14] In our case probably due to a super adder carcinomatous infiltration of GB and liver as well as rarity of such cases, CT scan could not bring out the diagnosis of Fasciolopsis to the forefront.

ERCP may demonstrate the adult worm within the CBD or gallbladder, can obtain ova in biliary and duodenal aspirate, and aids in the management by sphincterotomy and removal of the adult worms.[15] In our case, similarly, ERCP proved to be the best modality.

Although triclabendazole, efficient against both mature and immature Fasciola worms, is the drug of choice, it is not available in India.[14] The antiparasitic drugs used for the medical treatment of fascioliasis in the past (e.g., parenteral dehydroemetine and oral bithionol and praziquantel) have not been proven very effective.[12] Noting the inoperable nature of her carcinoma along with albendazole for parasitic cause, other symptomatic treatment, biliary cannulation with stenting, was done as a palliative measure.


   Conclusion Top


However, keeping in mind illiteracy, the low socioeconomic status of majority of the population, and close proximity to livestock, it should be kept as one of the differential diagnoses of abdominal pain if it is associated with eosinophilia. This case paves the way for extensive research to establish the causal association of F. hepatica with gallbladder carcinoma.

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

This study was financially supported by S 'O' A Deemed to be University, Kalinga Nagar, Bhubaneswar, Odisha.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

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Price TA, Tuazon CU, Simon GL. Fascioliasis: Case reports and review. Clin Infect Dis 1993;17:426-30.  Back to cited text no. 1
    
2.
Shastry A, Bhat S. Essentials of Medical Parasitology. 2nd ed., Ch. 11. New Delhi: Jaypee Publishers; 2019. p. 162-4.  Back to cited text no. 2
    
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Ghildiyal JP, Singh DP, Goyal RK. Cutaneous images of interest of fascioliasis from India. Indian J Pathol Microbiol 2014;57:499-500.  Back to cited text no. 5
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Marcos LA, Terashima A, Gotuzzo E. Update on hepatobiliary flukes: Fascioliasis, opisthorchiasis and clonorchiasis. Curr Opin Infect Dis 2008;21:523-30.  Back to cited text no. 6
    
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Roig GV. Hepatic fascioliasis in the Americas: A new challenge for therapeutic endoscopy. Gastrointest Endosc 2002;56:315-7.  Back to cited text no. 7
    
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Machicado C, Machicado JD, Maco V, Terashima A, Marcos LA. Association of Fasciola hepatica infection with liver fibrosis, cirrhosis, and cancer: A systematic review. PLoS Negl Trop Dis 2016;10:e0004962.  Back to cited text no. 8
    
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Jedlina L, Kozak-Ljunggren M, Wedrychowicz H. In vivo studies of the early, peritoneal, cellular and free radical response in rats infected with Fasciola hepatica by flow cytometric analysis. Exp Parasitol 2011;128:291-7.  Back to cited text no. 9
    
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Marcos LA, Yi P, Machicado A, Andrade R, Samalvides F, Sánchez J, et al. Hepatic fibrosis and Fasciola hepatica infection in cattle. J Helminthol 2007;81:381-6.  Back to cited text no. 10
    
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Hundal R, Shaffer EA. Gallbladder cancer: Epidemiology and outcome. Clin Epidemiol 2014;6:99-109.  Back to cited text no. 11
    
12.
Mannstadt M, Sing A, Leitritz L, Brenner-Maucher K, Bogner J. Conservative management of biliary obstruction due to Fasciola hepatica. Clin Infect Dis 2000;31:1301-3.  Back to cited text no. 12
    
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Nyindo M, Lukambagire AH. Fascioliasis: An ongoing zoonotic trematode infection. Bio Med Res Int 2015;2015:786195.  Back to cited text no. 13
    
14.
Ramanan RV, Dhus U, Ramamurthy A, Parameswaran SA, Piramanayagam P, Gopalakrishnan R. Human fascioliasis: Diagnosis by typical computed tomography features and response to nitazoxanide in 16 patients from India. Trop Gastroenterol 2018;39:149-54.  Back to cited text no. 14
    
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Gulsen MT, Savas MC, Koruk M, Kadayifci A, Demirci F. Fascioliasis: A report of five cases presenting with common bile duct obstruction. Neth J Med 2006;64:17-9.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2]



 

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