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Obstructive Jaundice Caused by Mucinous Cystic Tumor of Gallbladder: A Case Report and Literature Review

  • Sulai Liu1,2,3,#,
  • Zhihua Zhang1,2,3,#,
  • Chao Guo1,2,3,
  • Zhangtao Yu1,2,3,
  • Siyuan He1,2,3,
  • Junaid Khan1,2,3,
  • Bo Jiang1,2,3,
  • Yinghui Song1,2,3,* and
  • Chuang Peng1,2,3,*
 Author information
Journal of Clinical and Translational Hepatology   2021;9(4):598-602

doi: 10.14218/JCTH.2020.00123


Mucinous cystic tumor of the gallbladder is an extremely rare benign tumor, with potential for malignant degeneration. Mucinous cystic tumors of the cystic duct are divided into mucinous cystadenoma and mucinous cystadenocarcinoma. Currently, cystadenoma is generally considered to be a precancerous lesion of cystadenocarcinoma. At present, there are few cases reported worldwide, and there are no relevant guidelines for diagnosis and treatment of this disease. This article presents the collected clinical data of a patient with mucinous cystic tumor of the gallbladder who was admitted to the First Affiliated Hospital of Hunan Normal University, with the characteristics of the disease summarized in combination with a focused literature review.


Mucinous cystic tumor, Jaundice, Gallbladder, Case report, Literature review


The cystic duct mucinous cystic tumor is a rare cystic duct tumor with latent malignant lesions, and represents a special pathological type in cholangiocarcinoma.1,2 Mucinous cystic tumors of the cystic duct are divided into mucinous cystadenoma and mucinous cystadenocarcinoma.3 Currently, cystadenoma is generally considered to be a precancerous lesion of cystadenocarcinoma, so patients in whom this disease is suspected should be decisively operated, with their intraoperative fast frozen sections used to guide the operation.2,3 The etiology of mucinous cystic gland tumors of the bile ducts is currently unclear. Some believe that this is a congenital disease, caused by fluid retention as a result of inflammatory hyperplasia or obstruction of some abnormal ducts that occurs during embryonic growth; others believe that mucinous cystic tumors of the cystic duct are related to preembryonic intestinal residual or ectopic ovarian tissue.1–3

The patient presented with painless jaundice that had lasted for a duration of 1 month. In this case, the large cystic duct tumor was found to have squeezed the common bile duct to cause obstructive jaundice and dilated intrahepatic and extrahepatic bile ducts.

Case report

The patient was a 57 year old female, with complaint nausea and vomiting for more than 1 month. She had been treated at a local community hospital 1 month prior to presentation at our hospital. However, after having received intravenous fluids and antibiotics, her symptoms did not alleviate. Laboratory examination upon presentation to our hospital showed the following: total bilirubin of 143.9 μmol/L; direct bilirubin of 109.6 μmol/L; alanine aminotransferase of 87.7 U/L; aspartate aminotransferase of 78.25 U/L; alkaline phosphatase of 201 U/L; gamma-glutamyltransferase of 682.0 U/L; carbohydrate antigen 19–9 of 252.02 U/mL; cancer antigen 72–4 of 7.76 U/mL; and, negativity for the panel of antinuclear antibodies. Findings for cancer antigen 125, blood routine, and serum C-reactive protein were basically normal. Abdominal computed tomography (Fig. 1A, B) showed obstruction of the lower part of the common bile duct, dilatation of the upper bile duct, and chronic cholecystitis. Magnetic resonance choliangiopancreatography (Fig. 1C) showed thickening of the lower part of the common hepatic duct with dilatation of the bile ducts inside and outside the liver. For preoperative jaundice reduction and cholangiography, percutaneous transhepatic cholangial drainage (referred to as PTCD) was performed. The PTCD angiography (Fig. 1D) showed filling defect in the common bile duct and bile duct dilation.

Preoperative imaging revealed biliary obstruction.
Fig. 1  Preoperative imaging revealed biliary obstruction.

(A, B) Abdominal plain computed tomography scan plus enhanced (C) Magnetic resonance choliangiopancreatography and (D) PTCD angiography showed both intrahepatic bile duct dilation and common hepatic duct dilation, as well as a space-occupying lesion at the confluence of the cystic duct.

In order to clarify the nature of the space occupied by the bile duct and relieve the patient’s biliary obstruction, abdominal cavity exploration, biliary exploration, preparation of biliary and enteral drainage were performed. A frozen section was assessed intraoperatively, and the results were reported as: “Consider gallbladder cyst adenoma”. We then performed an open cholecystectomy, biliary exploration, and bile duct repair with shaping and T-tube drainage.

During the operation, a 4.0×2.0 cm mass of the cystic duct protruded into the bile duct lumen (Fig. 2A–D). The mass grew on the wall of the cystic duct with a pedicle, and a few stones were seen in the gallbladder.

A cystic duct-origin mass was found during the operation to block the common bile duct.
Fig. 2  A cystic duct-origin mass was found during the operation to block the common bile duct.

(A) 4.0×2.0 cm mass of the cystic duct was seen protruding into the bile duct cavity (white arrow). (B) The mass was found on the wall of the cystic duct (white arrow). (C) The upper common hepatic duct (white arrow) and the lower common bile duct (green arrow) did not show stenosis nor any masses. (D) A cystic duct-origin mass was observed.

The predischarge inspection showed that the total bilirubin was 25.8 μmol/L and the direct bilirubin was 18 μmol/L. Computed tomography of the abdomen showed that the dilatation of the bile ducts, inside and outside the liver, was significantly less than before. Postoperative pathology showed that there was a multicystic mass in the cystic duct, 4×2×2 cm in size, multicystic at the cut surface, and containing light-yellow, clear liquid in the cyst. The pathological diagnosis was mucinous cystic tumor with mild atypical hyperplasia with chronic cholecystitis immunohistochemistry of cytokeratin 7 (+), cytokeratin 19 (epithelial +), estrogen receptor (+), progesterone receptor (+), P53 (−), and Ki67 (scattered +) (Fig. 3).

Postoperative pathology showed that there was a multicystic mass in the cystic duct.
Fig. 3  Postoperative pathology showed that there was a multicystic mass in the cystic duct.

(A) A 100× cyst, lined with a single layer of mucin-producing epithelial cells and showing low-grade dysplasia was observed. (B) Most segments of the 400× cyst wall contained ovarian-like stroma. (C, D) 40× ovarian-like stroma immunohistochemical analysis showed positivity for estrogen receptor (ER) and progesterone receptor (PR).

Informed consent

Prior written informed consent was provided from the patient and this study was approved by the Ethics Review Board of Hunan Provincial People’s Hospital/The First Affiliated Hospital of Hunan Normal University.


Mucinous cystic neoplasms (MCNs) were first reported in pancreatic tissue and, subsequently, there has been much research devoted to investigating pancreatic MCNs. However, there are still many controversies about pancreatic MCN disease and even less is known about gallbladder MCN. According to the authors’ search of the PubMed database, the earliest case of gallbladder MCN was reported by Bishop in The Lancet in 1901,1 and there have been 16 literature reports on gallbladder MCN (Table 1).

Table 1

Gallbladder MCN reported cases

CaseYearAgeSexJaundiceAbdominal painTumor size in cmCarbohydrate antigen 19-9Reference
1190142FemaleYNullSize of a child’s headNull7
13201033FemaleNY0.67 × 0.28Null19
16201870FemaleNN6.7×6.8 ×7.2High2
18202057FemaleYN4.0 x 2.0HighCurrent study

Similar to pancreatic MCN, gallbladder MCN can manifest unilocular or multilocular cystic changes, containing septa. In the World Health Organization Classification of Digestive System Tumors (2010 Edition), biliary MCN is listed separately, as a special tumor of the gallbladder, and is classified into “mucocystic tumors with low-grade or medium-grade epithelium according to the status of intraepithelial neoplasia. Internal neoplasia (8470/0) (8470/2)”, “Invasive mucocystic carcinoma (8470/3)”. The existing literature data divides MCN into at least two types.1 One is non-invasive and has ovarian-like stroma under the epithelium, which is characterized by a high cell density. It appears as a dense collection of spindle-shaped cells lacking cytoplasm and is immune to estrogen and progesterone receptors. This subtype affects middle-aged women. The other type is more aggressive, has no ovarian-like stroma, and affects men between 75 and 88 years-old. There are others who classify MCN using three subtypes, based on epithelial atypia and infiltration; the subtypes are mucinous cyst-adenoma, non-invasive mucinous cystadenocarcinoma, and invasive mucinous cystadenocarcinoma.2

Both gallbladder MCN and pancreatic MCN are common in women. The difference is that pancreatic MCN often occurs in the body and tail of the pancreas, which do not often cause obstructive jaundice.3 In the case of gallbladder MCN, as the tumor increases, some patients will show painful or painless jaundice.4 The overall prognosis of the disease is good, but there is a certain malignant potential. According to a Japanese study encompassing 156 cases of pancreatic MCN resection, the 10-year survival rate after resection was 95% for adenoma and 63% for cancer, among which micro-invasive carcinoma also reached more than 90%.4 Another study showed that the 5-year survival rate of untreated pancreatic MCN with invasive carcinoma was about 30% and the prognosis was poor.5 Such statistics are still lacking for gallbladder MCN. In pancreatic MCN, the maximum tumor diameter is an independent risk factor affecting malignant transformation, and the level of carbohydrate antigen 19–9 has greater diagnostic significance for male patients.3 In gallbladder MCN, as the tumor size increases, the likelihood of jaundice and malignancy increases together. In our case, the cystic duct tumor was large and it compressed the common bile duct, which then caused obstructive jaundice and intrahepatic bile duct dilation. Additionally, since gallstones were present, the case could have been misdiagnosed as common bile duct stones or Mirizzi syndrome.

Therefore, preoperative examination is particularly important. For this disease, ultrasound is more sensitive to the internal features of the tumor (i.e. separation and fragments) and should be the first choice. Computed tomography can determine the location of the tumor and whether there is infiltration of surrounding tissues, which can help guide the scope of surgical resection. Magnetic resonance choliangiopancreatography can help determine the bile duct compression and involvement, determine the cause of jaundice in patients, and determine whether biliary reconstruction surgery is appropriate.6 Assessment of a quick-frozen section during the operation will help guard against the possibility of malignancy.

For asymptomatic patients, such as those who have tumors found on physical examination or imaging, one might use the pancreatic MCN endoscopic ultrasound-fine needle aspiration data on fluid collection to evaluate glucose (sensitivity of 92%, specificity of 87%, accuracy of 90%) and carcinoembryonic antigen (sensitivity of 58%, specificity of 96%, accuracy of 69%), for evaluation before an invasive operation, since there is always risk of tumor dissemination and surgical complications.3 It is important to comprehensively consider the patient’s sex, age, family history, and surgical conditions. Interestingly, almost all gallbladder MCN patients are female1,7–21 (Table 1). In treatment, surgical resection is recommended for patients with clinical symptoms, such as abdominal pain, bloating, jaundice, or asymptomatic patients with gallbladder stones.22 It is important to send fast frozen sections during the operation to guide the operation method. After the surgical resection, it is recommended to check the confluence of the cystic duct, the wall of the gallbladder, and the common bile duct for other malignant tumors.

In summary, there is currently a lack of consistent evidence for the malignant potential of gallbladder MCN, and there is also a lack of guidelines or consensus in diagnosis and treatment. However, the consensus reached after we compiled the literature is that due to the potential malignancy of gallbladder MCN, early diagnosis of such diseases should be paid attention to in clinical work, surgical treatment should be actively performed, and changes should be made according to the rapid intraoperative pathological examination results. Operating or expanding the scope of surgery will likely improve the prognosis and reduce recurrence and malignant transformation.



mucinous cystic neoplasm


percutaneous transhepatic cholangial drainage


Data sharing statement

All data are available upon request.


This work was financially supported by following funds: Huxiang Youth Talent Support Program (Grant No. 2020RC3066); Postdoctoral Innovation Talents Project (Grant No. 2020RC2064); Hunan Provincial Natural Science Foundation of China (Grant No. 2019JJ50320/2020JJ5610); The Project of Improving the Diagnosis and Treatment Capacity of Hepatobiliary, Pancreas and Intestine Disease in Hunan Province (Xiangwei [2019] Grant No. 118).

Conflict of interest

The authors have no conflict of interests related to this publication.

Authors’ contributions

Patient management (CG), drafting of the manuscript (SL, ZZ, JK), statistical analysis (YS, SH), data collection (SL, ZZ, YS, ZY, CP, BJ), and revision of the manuscript for important intellectual content (YS,CP)


  1. Zevallos Quiroz JC, Jiménez Agüero R, Garmendi Irizar M, Ruiz Montesinos I, Comba Miranda JW. Mucinous cystic neoplasm of the gallbladder obstructing the common bile duct, a rare entity with a new name. Cir Esp 2014;92(8):567-569 View Article
  2. Sugawara S, Hirai I, Watanabe T, Tezuka K, Kimura W. A case of mucinous cystic neoplasm of the gallbladder. Clin J Gastroenterol 2018;11(5):428-432 View Article
  3. Li WL, Xu YD, Han X, Wu WC, Lou WH. Clinical analysis and prognosis factors of malignancy in the patients with mucinous cystic neoplasms of the pancreas. Zhonghua Wai Ke Za Zhi 2020;58(3):225-229 View Article
  4. Tada M, Koike K. Pancreatic tumor:progress in diagnosis and treatment. Topics: II. Intraductal papillary mucinous neoplasm of the pancreas (IPMN) /mucinous cystic neoplasm (MCN); 1. Symptoms and surveillance of IPMN and MCN. Nihon Naika Gakkai Zasshi 2012;101(1):51-56 View Article
  5. Yamao K, Yanagisawa A, Takahashi K, Kimura W, Doi R, Fukushima N, et al. Clinicopathological features and prognosis of mucinous cystic neoplasm with ovarian-type stroma: a multi-institutional study of the Japan pancreas society. Pancreas 2011;40(1):67-71 View Article
  6. Xu HX. Contrast-enhanced ultrasound in the biliary system: Potential uses and indications. World J Radiol 2009;1(1):37-44 View Article
  7. Bishop ES. An undescribed innocent (?) growth of the gall-bladder. Lancet 1901;158(4063):72-73 View Article
  8. Kordenat RA. Cystadenoma of the gallbladder report of a case. Wis Med J 1930;29:634-637
  9. Shambaugh P. Multilocular papillary cystadenoma of the gall bladder. The American Journal of Surgery 1933;22(2):229-231 View Article
  10. Ishak KG, Willis GW, Cummins SD, Bullock AA. Biliary cystadenoma and cystadenocarcinoma: report of 14 cases and review of the literature. Cancer 1977;39(1):322-338
  11. Simmons TC, Miller C, Pesigan AM, Lewin KJ. Cystadenoma of the gallbladder. Am J Gastroenterol 1989;84(11):1427-1430
  12. Devaney K, Goodman ZD, Ishak KG. Hepatobiliary cystadenoma and cystadenocarcinoma. A light microscopic and immunohistochemical study of 70 patients. Am J Surg Pathol 1994;18(11):1078-1091
  13. Spector SA, Fernandez VE, Vernon SE, Dunkin B, Livingstone AS. Gallbladder cystadenoma and common bile duct obstruction. Int J Gastrointest Cancer 2003;34(2-3):151-155 View Article
  14. Terada T, Takeuchi T, Taniguchi M. Hepatobiliary cystadenocarcinoma with cystadenoma elements of the gall bladder in an old man. Pathol Int 2003;53(11):790-795 View Article
  15. Rooney TB, Schofer JM, Stanley MD, Banks SL. Biliary cystadenoma of the gallbladder. AJR Am J Roentgenol 2005;185(6):1571-1572 View Article
  16. Waldmann J, Zielke A, Moll R, Schweinsberg TS, Rothmund M, Langer P. Cystadenocarcinoma of the gallbladder. J Hepatobiliary Pancreat Surg 2006;13(6):594-599 View Article
  17. McCague A, Rosen M, O’Malley K. Laparoscopic cholecystectomy of a polypoid gallbladder cystadenoma obstructing the common bile duct. Surg Laparosc Endosc Percutan Tech 2008;18(2):209-212 View Article
  18. Sistla SC, Sankar G, Basu D, Venkatesan B. Biliary cystadenocarcinoma of the gall bladder: a case report. J Med Case Rep 2009;3:75 View Article
  19. Gokalp G, Dusak A, Topal NB, Aker S. Cystadenoma originating from the gallbladder. J Ultrasound Med 2010;29(4):663-666 View Article
  20. Moussa M, Douard R, Marzouk I, Kort I, Mekni A. Biliary cystadenoma and cystadenocarcinoma of the gallbladder: A clinical review. Am Surg 2017;83(6):e186-188
  21. Rivero-Soto RJ, Hossein-Zadeh Z, Coleman J, Ahuja V. A mucinous cystic neoplasm originating from the gallbladder: A case report and literature review. Perm J 2019;23:18-077 View Article
  22. Arnaoutakis DJ, Kim Y, Pulitano C, Zaydfudim V, Squires MH, Kooby D, et al. Management of biliary cystic tumors: a multi-institutional analysis of a rare liver tumor. Ann Surg 2015;261(2):361-367 View Article
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Obstructive Jaundice Caused by Mucinous Cystic Tumor of Gallbladder: A Case Report and Literature Review

Sulai Liu, Zhihua Zhang, Chao Guo, Zhangtao Yu, Siyuan He, Junaid Khan, Bo Jiang, Yinghui Song, Chuang Peng
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