Abstract
Background
Biliary cystadenomas are very rare benign tumors which can transform into cystadenocarcinomas. The largest case series reported on 221 cases over 30 years from 10 HPB centers, i.e., about 7 cases per center per decade. The recommended treatment is liver resection. Enucleation of biliary cystadenomas has been done rarely. The purpose of the study was to determine the outcome of enucleation of these cysts, particularly the mortality rate and the recurrence rate.
Methods
A keyword search was done using OVID followed by a search of the bibliography of papers describing the enucleation of biliary cystadenomas. Of 45 articles obtained, 25 were retained. The main reasons for exclusion were non-English language and review articles.
Results
One hundred three patients in the 25 studies were treated with enucleation. Thirteen studies described prior treatments that had failed with resulting recurrence requiring re-treatment. The main indication for enucleation was large central cysts for which liver resection would be high risk. There were no postoperative deaths in patients treated by enucleation. Thirteen studies provided long-term follow-up in 40 patients, a substantial number given the rarity of the tumor. There were no recurrences or transformations to malignancy.
Conclusions
Enucleation seems to represent a reasonable treatment technique for BCA, especially when a large cystic lesion is located centrally and/or would require a large liver resection with significant loss of parenchyma.
Similar content being viewed by others
References
Devaney K, Goodman ZD, Ishak KG. Hepatobiliary cystadenoma and cystadenocarcinoma. A light microscopic and immunohistochemical study of 70 patients. Am J Surg Path 1994;18:1078-91.
Arnaoutakis DJ, Kim Y, Pulitano C, Zaydfudim V, Squires MH, Kooby D, Groeschl R, Alexandrescu S, Bauer TW, Bloomston M, Soares K, Marques H, Gamblin TC, Popescu I, Adams R, Nagorney D, Barroso E, Maithel SK, Crawford M Sandroussi C, Marsh W, Pawlik TM. Management of biliary cystic tumors: a multi-institutional analysis of a rare liver tumor. Ann Surg 2015;261:361-7.
Thomas KT, Welch D, Trueblood A, Sulur P, Wise P, Gorden DL, Chari RS, Wright Jr TK, Washington K, Pinson CW. Effective treatment of biliary cystadenoma. Ann Surg 2005;241:769-73.
Vogt DP, Henderson JM, Chmielewski E. Cystadenoma and cystadenocarcinoma of the liver: a single center experience. J Am Coll Surg 2005;200:727-33.
Kim JH. Pure Laparoscopic Transhepatic Enucleation of a Mucinous Cystic Neoplasm Abutting the Major Hepatic Veins at the Hepatocaval Confluence (with Video). J Gastrointest Surg 2018;22:557.
Pinson CW, Munson JL, Rossi RL, Braasch JW. Enucleation of intrahepatic biliary cystadenomas. Surg Gynec & Obst 1989;168:534-7.
Kloppers JC, Krige JEJ, Bernon M, Thomson SR. Nomenclature for neoplastic biliary cysts. South African Gastroent Rev 2016;1:219-23.
Simo KA, McKillop IH, Ahrens WA, Martinie JB, Iannitti DA, Sindram D. Invasive biliary mucinous cystic neoplasm: a review. HPB 2012;14:725-40.
Kishida N, Shinoda M, Masugi Y, Itano O, Fujii-Nishimura Y, Ueno A, Kitago M, Hibi T, Abe Y, Yagi H, Tanimoto A, Tanabe M, Sakamaoto M, Kitagawa Y. Cystic tumor of the liver without ovarian-like stroma or bile duct communication: two case reports and a review of the literature. World Surg Oncol 2014;12:229.
Brachet D, Mucci S, Desolneux G, Leboulanger G, Amie F, Pessaux P, Arnaud J-P. The simultaneous occurrence of mucinous cystadenomas in liver and pancreas. Europ J Gastroent Hepatol 2007;19:801-4.
Morris M, Anderson C, Drake L, Redfield S, Subramony C, Vanderlan W. Giant biliary cystadenoma. J Surg Case Rep 2012;2012:15.
Schoonbroodt D, Horsmans Y, Gigot JF, Rahier J, Geubel AP. Biliary cystadenoma of the liver with elevated CA 19-9. Liver 1994;14:320-2.
Dixon E, Sutherland FR, Burak K, McKinnon G, May G. Cystadenoma of the liver without mesenchymal stroma in a female following hormonal therapy for acne. HPB 2001;3:183-6.
Cahill CJ, Bailey ME, Smith MG. Mucinous cystadenomas of the liver. Clin Oncol 1982;8:171-7.
Chen YW, Li CH, Liu Z, Dong JH, Zhang WZ, Jiang K. Surgical management of biliary cystadenoma and cystadenocarcinoma of the liver. Genet Molec Res 2014;13:6383-90.
Daniels JA, Coad JE, Payne WD, Kosari K, Sielaff TD. Biliary cystadenomas: hormone receptor expression and clinical management. Dig Dis Sci 2006;51:623-8.
Dixon E, Sutherland FR, Mitchell P, McKinnon G, Nayak V. Cystadenomas of the liver: a spectrum of disease. Can J Surg 2001;44:371-6.
Emre A, Serin KR, Ozden I, Tekant Y, Bilge O, Alper A, Güllüoğlu M, Güven K.Intrahepatic biliary cystic neoplasms: Surgical results of 9 patients and literature review. World J Gastroent 2011;17:361-5.
Erdogan D, Kloek J, Lamers WH, et al. Mucinous cystadenomas in liver: management and origin. Digestive Surg 2010;27:19-23.
Fragulidis GP, Pantiora EV, Kontis EA, Primetis E, Polydorou A, Karvouni E, Polymeneas G. Biliary Mucinous Cystic Neoplasm of the Liver with Ovarian Stroma and Elevated Serum and Cystic Fluid Cancer Antigen 19-9 Levels. Cureus 2017;9:e1863.
Henson SW, Jr., Gray HK, Dockerty MB. Benign tumors of the liver. VI. Multilocular cystadenomas. Surg Gynec & Obst. 1957;104:551-4.
Ishak KG, Willis GW, Cummins SD, Bullock AA. Biliary cystadenoma and cystadenocarcinoma: report of 14 cases and review of the literature. Cancer 1977;39:322-38.
Lau WY, Chow CH, Leung ML. Total excision of mucinous biliary cystadenoma. Aust NZ J Surg 1990;60:226-8.
Martel G, Alsharif J, Aubin JM, Marginean C, Mimeault R, Fairfull-Smith RJ, Mohammad WM, Balaa FK. The management of hepatobiliary cystadenomas: lessons learned. HPB 2013;15:617-22.
Pillai SA, Velayutham V, Perumal S, et al. Biliary Cystadenomas: A Case for Complete Resection. HPB Surgery 2012;2012:1-6.
Ruiz-Tovar J, Lopez-Buenadicha A, Moreno-Caparros A, Vazquez-Garza JN. Surgical management of simple liver cysts. Cirugia y Cirujanos 2012;80:52-5.
Sang X, Sun Y, Mao Y, Yang Z, Lu X, Yang H, Xu H, Zhong S, Huang J. Hepatobiliary cystadenomas and cystadenocarcinomas: a report of 33 cases. Liver International 2011;31:1337-44.
Teoh AY, Ng SS, Lee KF, Lai PB. Biliary cystadenoma and other complicated cystic lesions of the liver: diagnostic and therapeutic challenges. World J SurG 2006;30:1560-6.
Treska V, Ferda J, Daum O, Liska V, Skalicky T, Bruha J. Intrahepatic biliary cystadenoma-diagnosis and treatment options. Turkish J Gastroent 2016;27:252-6.
Koffron A, Rao S, Ferrario M, Abecassis M. Intrahepatic biliary cystadenoma: role of cyst fluid analysis and surgical management in the laparoscopic era. Surgery 2004;136:926-36.
Soares KC, Arnaoutakis DJ, Kamel I, Anders R, Adams RB, Bauer TW, Pawlik TM. Cystic neoplasms of the liver: biliary cystadenoma and cystadenocarcinoma. J Am Coll Surg 2014;218:119-28.
Alper A, Ariogul O, Emre A, Uras A, Okten A. Treatment of liver hemangiomas by enucleation. Arch Surg1988;123:660-1.
Kloppers JC, Krige JE, Bernon MM, Burmeister S, Jonas E, Thomson SR, Bornman PC. Resection of biliary mucinous cystic neoplasms of the liver: a prospective cohort series of 13 consecutive patients. South African Surg 2018;56:41-4.
Acknowledgements
The authors acknowledge the advice of Dr. Sanjeev Bhalla of the Department of Radiology and Dr. Adeel Khan of the Department of Surgery, Washington University in Saint Louis, in composing figure 1 The authors also thank Dr. Deyali Chatterjee of the Department of Pathology, Washington University in Saint Louis for her help in creation of Figure 2.
Funding
Siteman Comprehensive Cancer Center and NCI Cancer Center Support Grant P30 CA091842.
Author information
Authors and Affiliations
Corresponding author
Additional information
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Strasberg, S.M., Chapman, W.C. Enucleation of Biliary Cystadenomas: a Review. J Gastrointest Surg 25, 2700–2706 (2021). https://doi.org/10.1007/s11605-021-05106-x
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s11605-021-05106-x