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UNUSUAL CASE
Year :   |  Volume :   |  Issue :   |  Page :
 

Ciliated foregut cyst of the gall bladder in the Calot's triangle: A case report with review of literature


1 Belle Vue Clinic, Kolkata, West Bengal, India
2 Department of Minimal Access Surgery, Belle Vue Clinic, Kolkata, West Bengal, India

Date of Submission10-Dec-2021
Date of Acceptance10-Jan-2022
Date of Web Publication17-May-2022

Correspondence Address:
Pallawi Priya,
Belle Vue Clinic, 9 and 10, Loudon Street, Kolkata, West Bengal
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jmas.jmas_379_21

  Abstract 


Ciliated foregut cysts (CFCs) are rare anomalies of the foregut epithelium. The common sites in the abdomen are liver and gall bladder. There are only 16 cases of CFC reported in the gall bladder to date. A 20-year-old girl presented with pain in the right upper abdomen. There was a radiological evidence of cystic lesion in the region of Calot's triangle. A differential diagnosis of either type 2 choledochal cyst or CFC was made. Intraoperative and histopathological findings were suggestive of CFC. CFC should be kept in mind as a rare differential diagnosis when evaluating cysts in the gall bladder fossa.


Keywords: Cholecystectomy, ciliated foregut cyst, developmental anomalies of the gall bladder



How to cite this URL:
Baig SJ, Priya P. Ciliated foregut cyst of the gall bladder in the Calot's triangle: A case report with review of literature. J Min Access Surg [Epub ahead of print] [cited 2022 Aug 8]. Available from: https://www.journalofmas.com/preprintarticle.asp?id=345445





  Introduction Top


Ciliated foregut cysts (CFCs) are embryonic anomalies of the foregut epithelium.[1] The primitive foregut cells differentiate into the respiratory tract, foregut and the hepatobiliary system. Rarely, there may be aberrant formation of cysts with respiratory type epithelium. They may be located above or below the diaphragm. In the abdomen, the common sites are the liver and the gall bladder.[1]

CFC can be asymptomatic and present as a radiological dilemma. The management is usually surgical, and the role of expectant treatment in established CFC is not clear.

There are only 16 published reports of CFC in relation to the gall bladder to the best of our knowledge. The technical details of the surgical excision in a CFC located within the Calot's triangle have been addressed only in one case report.

We report the 17th case of CFC in relation to the gall bladder and the second case of CFC in the Calot's triangle. We have also reviewed the literature on the subject.


  Case Report Top


A 20-year-old girl presented with pain in the right upper abdomen for which an ultrasonography (USG) was done. It reported a cyst arising from the common hepatic duct (CHD). A provisional diagnosis of type 2 choledochal cyst was given. Complete blood counts, liver function, amylase and lipase levels were normal. A contrast-enhanced computed tomography (CT) scan showed a 5-cm hypodense space-occupying lesion sandwiched between CHD, cystic duct and inferior surface of the liver (Calot's triangle). No communication was seen [Figure 1]a. The provisional diagnosis offered was CFC, and the differential diagnoses offered were type 2 choledochal cyst and duplex gall bladder.
Figure 1: (a) Computed tomography image showing GB (red arrow) and the cyst (blue arrow), (b) Intraoperative image with the view of GB (red arrow) and the cyst (blue arrow), (c) View of the medial margin of ciliated foregut cyst (blue arrow) and common hepatic duct (CHD) (yellow arrow) after anterior dissection, (d) Completed anterior and posterior dissection showing no communication of the cyst (blue arrow) with the CHD (yellow arrow), (e) Specimen showing GB (red arrow) and the cyst (blue arrow), (f) Ciliated columnar epithelium on HPE

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In view of the above findings, we planned a surgical excision. Laparoscopy with standard 4 cholecystectomy ports showed a cyst in the Calot's triangle [Figure 1]b. We started the posterior dissection with a hook on low-voltage setting and opened the posterior space till the body of the gall bladder. The anterior dissection was started on the right margin of the common bile duct (CBD) moving cephalad till the hilum to delineate the medial margin of the cyst. The cystic artery was identified during this step, clipped and divided. After that, blunt dissection was done to lift the cyst from the CHD [Figure 1]c. The cyst was now approached both from the anterior and posterior sides till the distal third of the gall bladder with the cyst was lifted off from the inferior surface of the liver showing no communication of the cyst with the liver [Figure 1]d. The critical view of safety was established. The cystic duct was clipped and divided. The entire gall bladder with the cyst was dissected off the liver, put in a bag and extracted from the umbilical port. The cyst needed to be aspirated to ease extraction which revealed mucoid material. Specimen [Figure 1]e was sent for histopathology. The operative site was carefully examined for any bile leak and oozing. The fascial closure was done, and the skin was closed with subcuticular stitches. Postoperatively, the patient recovered well and was discharged after 48 h. Histopathology confirmed the diagnosis of CFC [Figure 1]f.


  Discussion Top


CFC is a congenital anomaly that represents the remnant of the primitive foregut, with majority occurring above the diaphragm. While more than 100 cases of hepatic CFC have been reported,[2] it is extremely rare in relation to the gall bladder. Only 16 cases are reported in the literature to the best of our knowledge, mostly at the neck region of the gall bladder. Ours is the 17th case report.

These are usually radiological or intraoperative incidental findings. Occasionally, patients may present with pain. They are most commonly reported in middle-aged women. Our patient, however, was a young female.

USG appearance is a hypoechoic or hyperechoic unilocular cyst. The sizes reported in the literature vary from 1 to 4.5 cm. CT or magnetic resonance imaging (MRI) is helpful in making a diagnosis although it may be difficult to distinguish them from choledochal cyst, pancreatic pseudocyst, parasitic cyst, hepatic cystadenoma and duplex gall bladder. In our case, the sonologist had made a diagnosis of a choledochal cyst. This was later diagnosed provisionally as a CFC on CT scan indicating the importance of CT/MRI in diagnosing this condition. A magnetic resonance cholangiopancreatography in this case may have added to the diagnostic certainty but could not be done due to financial constraints.

Management is controversial but is usually surgical. Although there are no reports of malignancy in CFC of the gall bladder, metaplasia and squamous cell carcinoma have been reported in CFC of the liver.[3] With this background knowledge, it may be prudent to excise the CFC of the gall bladder as prophylaxis against cancer even in asymptomatic cases since the aetiopathogenesis of the two conditions is similar.

Technical details of such cases are unsurprisingly sparse. Therefore, certain key steps need to be reported. The step that we found useful was to perform the excision adhering to the principles of safe cholecystectomy and demonstrating the critical view of safety. Therefore, we started dissecting from the posterior approach. The communications of the cyst have never been reported except one letter in the journal of Virchows Archives 2005 by Koletsa et al.[4] Although other published case reports have not demonstrated any cyst-bile duct communications, it is important to be aware of this. We took special care while bluntly dissecting off the cyst from the CHD and the liver with a suction cannula after incising the peritoneum with a diathermy on low settings. We did not use intraoperative cholangiography because anatomical landmarks (Rouviere's sulcus, CBD, portal vein and hepatic artery pulsation) were clear and we could demonstrate the critical view of safety. Histology showed the inner lining of cuboidal ciliated epithelium typical of the respiratory tract. Cytokeratin staining with IHC showed positive CK7 and negative CK20. This is the typical histological picture of CFC as published in the literature.


  Conclusion Top


Cysts at the gall bladder fossa should be assessed by CT/MR/both to confirm the diagnosis. CFC should be kept in mind as a rare differential diagnosis. Surgical excision is usually reported even in asymptomatic cases despite the low evidence of malignant potential. Laparoscopic excision is feasible in CFC of the gall bladder.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Sharma S, Corn A, Kohli V, Wright HI, Sebastian A, Jabbour N. Ciliated hepatic foregut cyst: An increasingly diagnosed condition. Dig Dis Sci 2008;53:2818-21.  Back to cited text no. 1
    
2.
Ambe C, Gonzalez-Cuyar L, Farooqui S, Hanna N, Cunningham SC, Ambe C, et al. Ciliated hepatic foregut cyst: 103 cases in the world literature. Open J Pathol 2012;2:45-9.  Back to cited text no. 2
    
3.
Bishop KC, Perrino CM, Ruzinova MB, Brunt EM. Ciliated hepatic foregut cyst: A report of 6 cases and a review of the English literature. Diagn Pathol 2015;10:81.  Back to cited text no. 3
    
4.
Koletsa T, Tzioufa V, Michalopoulos A, Apostolidis S, Papadopoulos B, Hytiroglou P. Ciliated hepatic foregut cyst communicating with the gallbladder. Virchows Arch 2005;446:200-1.  Back to cited text no. 4
    


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2004 Journal of Minimal Access Surgery
Published by Wolters Kluwer - Medknow
Online since 15th August '04