Case Series
 
Extrahepatic intraductal biliary cystadenomas: Solving the mystery of “Idiopathic biliary obstruction”? A tertiary care institute experience
Sugi R. V. Subramaniam1, Sugumar Chidambaranathan2, Senthil Kumar3, Prabhakaran Raju4, Jeswanth Sathyanesan5, Ravichandran Palaniappan6
1Post Graduate Resident, Institute of Surgical Gastroenterology & Liver Transplant Govt. Stanley Medical College, Chennai, Tamil Nadu, India
2Professor, Institute of Surgical Gastroenterology & Liver Transplant, Govt. Stanley Medical College, Chennai, Tamil Nadu, India
3Assistant Professor, Institute of Surgical Gastroenterology & Liver Transplant, Govt. Stanley Medical College, Chennai, Tamil Nadu, India
4Assistant Professor, Institute of Surgical Gastroenterology & Liver Transplant, Chennai, Tamil Nadu, India
5Professor, Institute of Surgical Gastroenterology & Liver Transplant, Govt. Stanley Medical College, Chennai, Tamil Nadu, India
6Professor& Head of Department, Institute of Surgical Gastroenterology & Liver Transplant, Govt. Stanley Medical College, Chennai, Tamil Nadu, India

Article ID: 100078Z04SS2018
doi: 10.5348/100078Z04SS2018CS

Corresponding Author:
Dr. Sugi R. V. Subramaniam
601, Old Jail Road
Institute of Surgical Gastroenterology & Liver Transplant
Govt. Stanley Medical College, Chennai
Royapuram, Tamil Nadu 600001, India

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How to cite this article
Sugi R V, Sugumar C, Senthil Kumar P, Prabhakaran R, Jeswanth S, Ravichandran P. Extrahepatic intraductal biliary cystadenomas: Solving the mystery of “Idiopathic biliary obstruction”? A tertiary care institute experience. Int J Hepatobiliary Pancreat Dis 2018;8:100078Z04SS2018.


ABSTRACT

Introduction: Biliary cystadenoma is a rare benign neoplasm of the liver with less than 200 cases being reported all over the world. The greatest challenge in the management of biliary cystadenoma lies in the pre-operative diagnosis, since it is most often misdiagnosed as simple liver cyst or hydatid cyst. We report a series of 12 cases highlighting the radiological findings and problems related to its management with special focus on intrahepatic biliary cystadenomas; a very unusual benign cause of obstructive jaundice and their management.

Case Series: Records of 12 patients who underwent surgery for biliary cystadenomas, between 2013 and 2016, were reviewed and analysed retrospectively. Of the 12 patients with biliary cystadenomas, three patients had features of obstructive jaundice in the absence of any other recognized cause of biliary obstruction. Majority of the patients were females. The most frequent symptom was abdominal pain (92%); with obstructive jaundice seen in three patients (25%). All patients were subjected to routine blood investigations with Liver function tests and in addition Serum CA 19-9 was done. We had three patients who had protruding mass like lesion into the biliary tract causing obstruction to bile flow. MRI with MRCP was done in all patients before subjecting them to surgical exploration; who were managed with extrication of the growth along with enucleation/ resection. There has been no recurrence during the follow-up period ranging from six months to three years. All 12 patients underwent surgical management which included enucleation in four patients and non-anatomical liver resection in eight patients and some in addition required T-tube drainage of the bile duct. Totally 25 cases of Biliary Cystadenomas have been managed in our Institute till date with only three cases of Obstructive jaundice.

Conclusion: In patients with obstructive jaundice, especially middle aged women, with the background of normal CA 19-9 levels and a polypodal mass projecting into the bile duct lumen intraductal biliary cystadenomas should be considered as a possible diagnosis. If feasible limited resection of the cyst with extrication of this benign lesion with close follow up and frequent imaging of the liver is advisable to locate early recurrences if any.

Keywords: Biliary cystadenoma, Episodic jaundice, Obstructive jaundice


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Acknowledgements
Dr. Sastha Ahantha Pillai, Dr. J. Saravanan & Dr. Ashwin Rammohan
Author Contributions
Sugi R. V. Subramaniam – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Sugumar Chidambaranathan – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Senthil Kumar – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Prabhakaran Raju – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Jeswanth Sathyanesan – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Ravichandran Palaniappan – Substantial contributions to conception and design, Acquisition of data, Analysis and interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Guarantor of Submission
The corresponding author is the guarantor of submission.
Source of Support
None
Consent Statement
Written informed consent was obtained from the patient for publication of this case series.
Conflict of Interest
Author declares no conflict of interest.
Copyright
© 2018 Sugi R. V. Subramaniam et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.

About the Authors


Sugi R. V. Subramaniam is Post Graduate Resident at Institute of Surgical Gastroenterology & Liver Transplant ,Govt. Stanley Medical College, Chennai,Tamil Nadu, India. I earned from undergraduate degree (MBBS) from Madras Medical College, Chennai, India & my Post Graduate degree (MS in General Surgery) from JIPMER, Puducherry, India. I am currently pursuing my Super Speciality (M.Ch in Surgical Gastroenterology) at Institute of Surgical Gastroenterology & Liver Transplant,Govt. Stanley Medical College,Chennai, Tamil Nadu, India. My field of Interest is Heaptobiliaryand Liver/Pancreas Transplantation. I have published few case reports and original articles. Recently reviewed an article for Surgical Innovation. https://publons.com/author/1519279/sugi-r-v#profile
Email: drrvsugi@gmail.com



Sugumar Chidambaranathan is Professor Institute of Surgical Gastroenterology & Liver Transplant Govt. Stanley Medical College Chennai, Tamil Nadu, India.
Email: dr.sugumar@yahoo.com



Senthil Kumar is Senior Assistant Professor, Institute of Surgical Gastroenterology & Liver Transplant Govt. Stanley Medical College, Chennai, Tamil Nadu, India.
Email: dr.psenthil@yahoo.com



Prabhakaran Raju is Senior Assistant Professor, Institute of Surgical Gastroenterology & Liver Transplant, Chennai, Tamil Nadu, India.
Email: kalprabha@gmail.com



Jeswanth Sathyanesan is Professor, Institute of Surgical Gastroenterology & Liver Transplant, Govt. Stanley Medical College, Chennai, Tamil Nadu, India.
Email: dr_jeswanth@yahoo.co.in



Ravichandran Palaniappan is Professor & Head of Department, Institute of Surgical Gastroenterology & Liver Transplant, Govt. Stanley Medical College, Chennai, Tamil Nadu, India. Email: prahari05@yahoo.com