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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3853860/?report=printable

J Indian Assoc Pediatr Surg. 2013 Oct-Dec; 18(4): 158159.

doi: 10.4103/0971-9261.121128

PMCID: PMC3853860

Hydatid cyst within a choledochal cystRuchirendu Sarkar, Ram Mohan Shukla, Sujay Maitra, Malay Bhattacharya, and Biswanath Mukhopadhyay

Department of Pediatric Surgery, Nil Ratan Sircar Medical College and Hospital, Kolkata, West Bengal, India

Address for correspondence:Dr. Ram Mohan Shukla, 7E, Dinobandhu Mukherjee Lane, Sibpur , How rah - 711 102, West Bengal, India. E-

mail: rammohanshukla@yahoo.co.in

Copyright: Journal of Indian Association of Pediatric Surgeons

This is an open-acces s article distributed under the terms of the Creative Commons A ttribution-Noncommercial-Share Alike 3.0 Unported, w hich

permits unrestricted use, distribution, and reproduction in any medium, provided theoriginal w ork is properly cited.

Abstract

A 5 year 4 months old male child presenting with pain abdomen and jaundice was diagnosed to havetype 1 choledochal cyst on ultrasonography and magnetic resonance cholangio pancreatography. On

exploration, the cystic dilatation of common bile duct was found to have a hydatid cyst (HC) inside it.

The per-operative findings were confirmed by histopathology. Association of HC within a choledochal

cyst is extremely rare and has been reported only twice before in the available English literature.

KEY WORDS: Choledochal cyst, hepaticodochoduodenostomy, hydatid cyst

INTRODUCTION

Hydatid cyst (HC) is a common parasitic disease in the Indian subcontinent. It commonly involves the

liver and lung, but other uncommon locations have been described in the literature.[1] Association of

HC within a choledochal cyst is extremely rare and has been reported only twice before in the available

English literature.

CASE REPORT

A child aged 5 years 4 months presented to our institute with the chief complaint of repeated attacks of

pain over right upper abdomen for last 4 months. Parents noticed gradually increasing yellow

discoloration of eyes for last 15 days. The child also had generalized itching all over the body and he was

passing deep yellow colored urine and clay colored stool for the same duration. On clinical examination,

patient was found to have deep jaundice. Epigastric tenderness was a prominent feature.

The child was admitted and investigated. The liver function tests were found to be grossly abnormalwith high total and conjugated bilirubin. Prothrombin time was initially abnormal and came down to

normal with treatment. Ultrasonography showed a dilatation of common bile duct (CBD) suggesting

choledochal cyst [Figure 1]. Magnetic resonance cholangio pancreatography confirmed the diagnosis [

Figure 2]. On exploration, the CBD was found to be dilated with a maximum diameter of about 3.5 cm.

After dissection, choledochotomy was done and it revealed a cyst with yellowish white membrane

suggesting bile-stained HC [Figure 3]. Extraction of the HC was performed followed by cholecystectomy

and complete excision of the choledochal cyst with hepaticodochoduodenostomy. Post-operatively both

resected specimen and the cyst within the CBD were sent for histopathological examination (HPE). The

results of histopathology were confirmatory. Post-operative hippuric iminodiacetic acid scan showed

normal flow of dye through hepaticodochoduodenostomy.

http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3853860/figure/F2/http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3853860/figure/F1/http://www.ncbi.nlm.nih.gov/pmc/about/copyright.htmlhttp://www.ncbi.nlm.nih.gov/pmc/about/copyright.htmlmailto:rammohanshukla@yahoo.co.inhttp://www.ncbi.nlm.nih.gov/pubmed/?term=Mukhopadhyay%20B%5Bauth%5Dhttp://www.ncbi.nlm.nih.gov/pubmed/?term=Bhattacharya%20M%5Bauth%5Dhttp://www.ncbi.nlm.nih.gov/pubmed/?term=Maitra%20S%5Bauth%5Dhttp://www.ncbi.nlm.nih.gov/pubmed/?term=Shukla%20RM%5Bauth%5Dhttp://www.ncbi.nlm.nih.gov/pubmed/?term=Sarkar%20R%5Bauth%5Dhttp://www.ncbi.nlm.nih.gov/pmc/articles/PMC3853860/figure/F3/http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3853860/figure/F2/http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3853860/figure/F1/http://dx.doi.org/10.4103%2F0971-9261.121128

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DISCUSSION

Pediatric HC disease commonly involves the lung and liver, but unusual locations like within the CBD,

head of pancreas etc. have been described in children.[1,2,3,4] Most of the cases of HC are

asymptomatic. However when symptomatic, the usual presentation is with pain in right

hypochondrium with obstructive jaundice due to obstruction of the biliary tree. Our case is a very rare

combination of both the pathologies presenting together.

The differential diagnosis includes pancreatic HC, which can also mimic choledochal cyst leading to

misdiagnosis as mentioned in the literature.[5,6]

Here, we discuss a very rare presentation of pediatric HC within a choledochal cyst type I variant. A

similar case was described by Gopal in 1993[7] where the authors did choledochocystoduodenostomy

because of dense adhesions around the cyst. However in our case, cholecystectomy along with complete

cyst excision, hepaticodochoduodenostomy[8] and removal of HC was done, which is regarded as one of

the standard approaches.

As per the editorial comment in the above article[7] the possibility that there was a small, primary

intrahepatic cyst from which a small daughter cyst traversed the biliary system and got lodged in the

CBD causing its gradual dilatation also came to our mind. To rule out this possibility, we sent the

specimen of the choledochal cyst and the HC for detailed HPE. On HPE, it was confirmed that there

was already a choledochal cyst, in which the HC (3.5 cm in its greatest axis) developed later making this

case very unique and extremely rare. Patient had an uneventful post-operative period.

In our case, it is possible that the migration of embryos occurred via the portal circulation to the liver

and then these embryos got lodged into the already formed choledochal cyst. The presence of the double

pathology made the diagnosis difficult in our patient.

CONCLUSION

HC may lodge within a choledochal cyst also, which is a very unusual location for it. Though very

unusual, it is very important to keep it in mind during the surgery of any child presenting to us with

obstructive jaundice and pain, to prevent rupture and dissemination of the HC, which usually isdiagnosed on the operating table in spite of all the relevant pre-operative investigations as seen in our

case.

Footnotes

Source of Support:Nil

Conflict of Interest:None declared.

REFERENCES

1. Dagtekin A, Koseoglu A, Kara E, Karabag H, Avci E, Torun F, et al. Unusual location of hydatid cysts

in pediatric patients. Pediatr Neurosurg. 2009;45:37983. [PubMed: 19940536]

2. De U, Basu M. Hydatid cyst of common bile duct mimicking type 1 choledochal cyst. J Indian Assoc

Pediatr Surg. 2007;12:834.

3. Gangopadhyay AN, Sahoo SP, Sharma SP, Gupta DK, Sinha CK, Rai SN. Hydatid disease in children

may have an atypical presentation. Pediatr Surg I nt. 2000;16:8990. [PubMed: 10663846]

4. Otgn I, Karnak I, Haliloglu M, Senocak ME. Obstructive jaundice caused by primary choledochal

hydatid cyst mimicking radiologically choledochal cyst. J Pediatr Surg. 2003;38:2568.

[PubMed: 12596118]

5. Mandelia A, Wahal A, Solanki S, Srinivas M, Bhatnagar V. Pancreatic hydatid cyst masquerading as a

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choledochal cyst. J Pediatr Surg. 2012;47:e414. [PubMed: 23164030]

6. Bhat NA, Rashid KA, Wani I, Wani S, Syeed A. Hydatid cyst of the pancreas mimicking choledochal

cyst. Ann Saudi Med. 2011;31:5368. [PMCID: PMC3183692] [PubMed: 21911995]

7. Gopal SC, Gangopadhyay AN, Gupta A. Children presenting with hydatid cysts in common bile duct

and choledochal cyst. Pediatr Surg I nt. 1993;8:1257.

8. Mukhopadhyay B, Shukla RM, Mukhopadhyay M, Mandal KC, Mukherjee PP, Roy D, et al.

Choledochal cyst: A review of 79 cases and the role of hepaticodochoduodenostomy. J Indian Assoc

Pediatr Surg. 2011;16:547. [PMCID: PMC3119937] [PubMed: 21731232]

Figures and Tables

Figure 1

Ultrasonography showing cystic dilatation of the co mmon bile duct

Figure 2

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Magnetic resonance cholangiopancreatography showing type 1 choledoc hal cyst

Figure 3

Choledocho tomy showing hydatid cyst within choledoc hal cyst

Articles from Journal of Indian Association of Pediatric Surgeons are provided here courtesy of Medknow Publications