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Year : 2018  |  Volume : 8  |  Issue : 2  |  Page : 114-117  

Isolated hydatid cyst of spleen with cystocolic fistula: An unusual case


1 Department of General Surgery, Kalpana Chawla Government Medical College, Karnal, Haryana, India
2 Department of Radiology, Kalpana Chawla Government Medical College, Karnal, Haryana, India

Date of Web Publication27-Dec-2018

Correspondence Address:
Ravi Kumar Garg
Department of General Surgery, Kalpana Chawla Government Medical College, Room No. 309, 3rd Floor, OPD Block, Karnal, Haryana
India
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DOI: 10.4103/tp.TP_63_17

PMID: 30693220

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   Abstract 


Hydatid cyst is a zoonotic disease caused by the cestode Echinococcus granulosus. Humans are incidental intermediate host. Liver and lungs are most common sites involved in hydatid cyst. Spleen, kidney, central nervous system, thyroid, breast are other rare sites. Here we present a unique case of a 48 yr female having isolated hydatid cyst of spleen, with the cyst having ruptured into the splenic flexure of colon.

Keywords: Cystocolonic fistula, Echinococcus, hydatid cyst, spleen, zoonosis


How to cite this article:
Garg RK, Katyal A, Bishnoi SR, Dahiya A. Isolated hydatid cyst of spleen with cystocolic fistula: An unusual case. Trop Parasitol 2018;8:114-7

How to cite this URL:
Garg RK, Katyal A, Bishnoi SR, Dahiya A. Isolated hydatid cyst of spleen with cystocolic fistula: An unusual case. Trop Parasitol [serial online] 2018 [cited 2019 Nov 15];8:114-7. Available from: http://www.tropicalparasitology.org/text.asp?2018/8/2/114/248693




   Introduction Top


Hydatid disease is a zoonotic disease where dogs and wolves are definitive hosts. Intermediate hosts such as cattle and sheep get infected when they consume grass or vegetables which are contaminated with eggs passed in faeces by definitive hosts.[1] Humans are the incidental intermediate hosts. Liver and lungs are the most common sites for hydatid cysts since they act as a primary filter for these oncospheres.[2] The spleen, kidney, bone, thyroid, and pancreas are some other organs involved in hydatid disease.[3] The incidence of splenic hydatid cyst is <5% even in endemic areas.[4] Here, we report an intriguing case of isolated hydatid splenic cyst which had formed a fistula in the splenic flexure of the colon.


   Case Report Top


A 48-year-old female presented in our outpatient department with on-and-off colicky pain in the left hypochondrium along with loose stools, emaciation, and on-and-off fever with chills for the last 1½ years. On clinical examination, there was pallor, pulse rate was 120/min, oral temperature was 100.2°F, and blood pressure was 110/70 mmHg. On palpation, abdomen was soft with mild tenderness in the left hypochondrium, with no definitive palpable mass. Blood investigations revealed hemoglobin: 5.4 g/dl, platelet count: 1.76 million/mm3, total leukocyte count: 17600/mm3, and differential leukocyte count was predominantly neutrophilic (85%). The patient's renal function tests, fasting blood sugar levels, and liver function tests were normal. The hydatid serology (immunoglobulin G antibodies, ELISA) of the patient was positive.

A computed tomography scan done 2 months back at some other infirmary revealed a large air-filled cystic lesion with a rim of peripheral calcification in the left upper quadrant [Figure 1]. Since it had no thoracic communication, thus the most likely source of air was its communication with the bowel.
Figure 1: Initial computed tomography scan of the patient, axial sections showing air-filled splenic hydatid cyst with thick rim of peripheral calcification. A small fluid level is seen in image (d) A small fluid level is seen in image shown by white arrow

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At our institution, an X-ray abdomen (erect, anteroposterior [AP] view) was done. It showed a radiolucent area in the left upper quadrant with membrane-like densities [Figure 2]a. Subsequently, the patient underwent a contrast-enhanced computed tomography abdomen. It showed a large, well-defined, rounded cystic lesion measuring 11.4 cm × 9.2 cm × 10.8 cm approximately (AP × TRA × CC) in the inferior aspect of spleen with no appreciable enhancement but with air-fluid level, wall calcifications, and calcified intraluminal membranes [Figure 2]b. There was also a large exophytic component with loss of fat planes with splenic flexure of transverse colon and lateral abdominal wall [Figure 3]. The involved splenic flexure had thickened walls (wall thickness: 6.8 mm). On administration of diluted nonionic contrast per rectum, the contrast was found to accumulate within the cyst [Figure 4]. A nonenhancing, wedge-shaped, hypodense area was also noted in the upper pole of the spleen (7.2 cm × 3.4 cm) suggestive of an infarct [Figure 3]a. The left kidney was shrunken and pushed posteriorly by the cyst [Figure 3]b. The patient had mild hepatomegaly and no other abnormality was found in the liver and lungs.
Figure 2: (a) Digital scout image obtained from second abdominal computed tomography 2 months later shows lucent (air-filled) cyst in the left upper quadrant with membranous densities within it representing detachment of endocyst from the pericyst (solid arrow). Corresponding coronal computed tomography image (b) clearly showing the collapsed membranes and wall calcifications. Adherent splenic flexure of colon noted in the inferior aspect shown by dotted arrows

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Figure 3: Axial and coronal images obtained pre (a and c) and post (b and d) diluted rectal nonionic contrast administration demonstrate contrast within the lesion and increase in the fluid level (dashed arrow). Rectal contrast is also seen in the colon (arrow, d). Splenic tissue is seen at the anterior and posterior periphery of the cyst in b (solid arrows)

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Figure 4: Axial postcontrast computed tomography images (a) Splenic infarct in upper pole of spleen with viable splenic tissue on its either side. (b) Dotted arrow showing adherent cyst wall to the parietal peritoneum with separation of the endocyst and pericyst. Shrunken and posteriorly displaced left kidney is also shown by solid arrow. (c) Loss of fat plane with pancreatic tail demonstrated by asterisk. (d) Wall thickening of adjacent splenic flexure of the colon (hollow arrow)

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About 4 units of packed red blood cells were given preoperatively to build up the hemoglobin of the patient. Vaccines for Streptococcus pneumonia, Haemophilus influenzae, and meningococcus were administered 2 weeks before surgery.

Intraoperatively, a large cystic lesion was seen over the convex surface of the spleen which was densely adherent to the lateral abdominal wall. At lower end, cyst was seen communicating with splenic flexure of colon. 10% povidone-iodine-soaked sponges were used to pack whole of abdominal cavity except small area over cyst. On aspiration of cyst, initially, free gas having foul fecal smell came out followed by faecal matter. Cavity was opened and its contents (faecal matter with peel of vegetables) were sucked out. No active daughter cyst and germinal layer were found due to calcification. Whole of the spleen along with calcified cyst and part of transverse colon were excised [Figure 5]. Colocolic primary anastomosis was made along with covering loop ileostomy.
Figure 5: Forceps tip passing from colonic end of fistula tract to hydatid cyst cavity

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The patient was kept nil per oral till stoma became functional. Thereafter, the patient was started orally and subsequently discharged with oral albendazole 10 mg/kg to be taken twice a day for 2 weeks. The patient is on regular follow-up and doing well. We plan to close the ileostomy after distal cologram to ensure that there is absolutely no leakage from colocolic anastomosis.


   Discussion Top


Hydatid cyst is caused by larvae of Echinococcus granulosus, Echinococcus multilocularis, and Echinococcus oligarthus.[5] Splenic hydatid cyst occurs when the oncospheres have passed through two filters of the liver and lungs and subsequently get trapped within the splenic sinusoids.[4] Some suggest retrograde venous spread from the liver to portal vein and then through splenic vein to spleen. As the cyst grows in size, it causes pressure effect on segmental arteries of the spleen causing infarction in its parts as seen in our case. Moreover, pressure over adjacent organs such as the stomach, pancreas, kidney, diaphragm, and inflammatory reaction of pericyst with these organs may cause dense adhesions and fistula formation. A potentially fatal complication is anaphylactic shock, which may occur due to spontaneous rupture or traumatic rupture of the cyst.

In our case, spontaneous rupture of cyst in the colon had led to colicky abdominal pain with chronic diarrhea, while the secondary infection of cyst cavity after fecal contamination had led to fever. Moreover, the enlarged cyst had caused compression over left renal artery, thereby causing shrinkage of the left kidney.

Various treatments have been described for hydatid cyst of liver[6] (e.g., total excision, removal of ectocyst and endocyst and cavity management with external tube drainage, omentoplasty, capsulorrhaphy, and capitonnage). However, for splenic hydatid cyst, splenectomy (total or partial) is the treatment of choice.[4]

In this patient, very dense adhesions were already present in splenic area, thus we had done primary anastomosis of colon instead of colostomy because a second surgery would have been difficult due to adhesions in the supracolic compartment. Therefore, we decided to go for covering ileostomy since the infracolic compartment would be free from adhesions in second surgery during stoma closure.

In our case, the history of fondling the stray dogs was present. Various risk factors for hydatid cyst in humans are the presence of large numbers of stray dogs harboring E. granulosus worms in that locality, inadequate facilities for slaughter, destruction of infected viscera of livestock, and access of dogs to infected offal.[7]

Health education, practice of handwashing after feeding and playing with dogs, proper disposal of infected viscera of livestock after slaughtering, and regular deworming of dogs, and persons who are at high risk for E. granulosus infection will decrease the incidence of hydatid cyst in human beings.

The most unique thing our case is that hitherto, only one incident of splenic hydatid cyst with colonic rupture has been reported in a 77-year-old female.[4] Our case is the second reported case in the literature of isolated hydatid cyst of the spleen with colonic fistula with left renal atrophy.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for images and other clinical information to be reported in the journal. The patient understands that name and initial will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Pedrosa I, Saíz A, Arrazola J, Ferreirós J, Pedrosa CS. Hydatid disease: Radiologic and pathologic features and complications. Radiographics 2000;20:795-817.  Back to cited text no. 1
    
2.
Bougioukas IG, Courcoutsakis N, Korakianitis OS, Tentes AA, Prasopoulos P. Liver hydatid cyst perforated into the large bowel: A case report. Cases J 2009;2:6999.  Back to cited text no. 2
    
3.
Wilkinson A, Mahore S, Chikhlikar K, Gohad G, Bothale K. Hydatid cysts presenting in uncommon sites: An uncommon case series. SEAJCRR 2014;3:645-56.  Back to cited text no. 3
    
4.
Teke Z, Yagci AB, Atalay AO, Kabay B. Splenic hydatid cyst perforating into the colon manifesting as acute massive lower gastrointestinal bleeding: An unusual presentation of disseminated abdominal echinococcosis. Singapore Med J 2008;49:e113-6.  Back to cited text no. 4
    
5.
Eckert J, Deplazes P. Biological, epidemiological, and clinical aspects of echinococcosis, a zoonosis of increasing concern. Clin Microbiol Rev 2004;17:107-35.  Back to cited text no. 5
    
6.
Sözüer EM, Ok E, Arslan M. The perforation problem in hydatid disease. Am J Trop Med Hyg 2002;66:575-7.  Back to cited text no. 6
    
7.
Acosta-Jamett G, Weitzel T, Boufana B, Adones C, Bahamonde A, Abarca K, et al. Prevalence and risk factors for echinococcal infection in a rural area of Northern Chile: A household-based cross-sectional study. PLoS Negl Trop Dis 2014;8:e3090.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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