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

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Obstructive Jaundice Caused by Mucinous Cystic Tumor of Gallbladder: a Case Report and Literature Review Journal of Clinical and Translational Hepatology 2021 vol. 000(000) | 000–000 Epub DOI: 10.14218/JCTH.2020.00123 Case Report 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* 1Department of Hepatobiliary Surgery/Hunan Research Center of Biliary Disease, Hunan Provincial People’s Hospital/The First Affiliated Hospital of Hunan Normal University, Changsha, Hunan, China; 2Biliary Disease Research Laboratory of Hunan Provincial People’s Hospital, Key Laboratory of Hunan Normal University, Changsha, Hunan, China; 3Clinical Medical Technology Research Center of Hunan Provincial for Biliary Disease Prevention and Treatment, Changsha, Hunan, China Received: 18 November 2020 | Revised: 3 January 2021 | Accepted: 19 March 2021 | Published: 00 Month 2021 Abstract 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 Mucinous cystic tumor of the gallbladder is an extremely a congenital disease, caused by fluid retention as a result of rare benign tumor, with potential for malignant degenera- inflammatory hyperplasia or obstruction of some abnormal tion. Mucinous cystic tumors of the cystic duct are divided ducts that occurs during embryonic growth; others believe into mucinous cystadenoma and mucinous cystadenocarci- that mucinous cystic tumors of the cystic duct are related to noma. Currently, cystadenoma is generally considered to be preembryonic intestinal residual or ectopic ovarian tissue.1–3 a precancerous lesion of cystadenocarcinoma. At present, The patient presented with painless jaundice that had there are few cases reported worldwide, and there are no lasted for a duration of 1 month. In this case, the large relevant guidelines for diagnosis and treatment of this dis- cystic duct tumor was found to have squeezed the common ease. This article presents the collected clinical data of a bile duct to cause obstructive jaundice and dilated intrahe- patient with mucinous cystic tumor of the gallbladder who patic and extrahepatic bile ducts. was admitted to the First Affiliated Hospital of Hunan Nor- mal University, with the characteristics of the disease sum- marized in combination with a focused literature review. Case report Citation of this article: Liu S, Zhang Z, Guo C, Yu Z, He S, Khan J, et al. Obstructive jaundice caused by muci- The patient was a 57 year old female, with complaint nausea nous cystic tumor of gallbladder: A case report and litera- and vomiting for more than 1 month. She had been treated ture review. J Clin Transl Hepatol 2021;00(00):00–00. doi: at a local community hospital 1 month prior to presentation 10.14218/JCTH.2020.00123. at our hospital. However, after having received intravenous fluids and antibiotics, her symptoms did not alleviate. Labo- ratory examination upon presentation to our hospital showed the following: total bilirubin of 143.9 μmol/L; direct bilirubin Introduction of 109.6 μmol/L; alanine aminotransferase of 87.7 U/L; as- partate aminotransferase of 78.25 U/L; alkaline phosphatase of 201 U/L; gamma-glutamyltransferase of 682.0 U/L; carbo- The cystic duct mucinous cystic tumor is a rare cystic duct hydrate antigen 19–9 of 252.02 U/mL; cancer antigen 72–4 tumor with latent malignant lesions, and represents a spe- 1,2 of 7.76 U/mL; and, negativity for the panel of antinuclear cial pathological type in cholangiocarcinoma. Mucinous antibodies. Findings for cancer antigen 125, blood routine, cystic tumors of the cystic duct are divided into mucinous 3 and serum C-reactive protein were basically normal. Abdomi- cystadenoma and mucinous cystadenocarcinoma. Cur- nal computed tomography (Fig. 1A, B) showed obstruction of rently, cystadenoma is generally considered to be a precan- the lower part of the common bile duct, dilatation of the up- cerous lesion of cystadenocarcinoma, so patients in whom per bile duct, and chronic cholecystitis. Magnetic resonance this disease is suspected should be decisively operated, with 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 preopera- Keywords: Mucinous cystic tumor; Jaundice; Gallbladder; Xase report; Litera- tive jaundice reduction and cholangiography, percutaneous ture review. transhepatic cholangial drainage (referred to as PTCD) was Abbreviations: MCN, mucinous cystic neoplasm; PTCD, percutaneous transhe- patic cholangial drainage. performed. The PTCD angiography (Fig. 1D) showed filling #These authors contributed equally to this work. defect in the common bile duct and bile duct dilation. *Correspondence to: Yinghui Song and Chuang Peng, Department of Hepato- In order to clarify the nature of the space occupied by biliary Surgery, Hunan Provincial People’s Hospital/The First Affiliated Hospital the bile duct and relieve the patient’s biliary obstruction, of Hunan Normal University, Changsha, Hunan 410004, China. Tel: +86-731- 83929520, Fax: +86-731-83929520, E-mail: [email protected] (YS) or peng- abdominal cavity exploration, biliary exploration, prepara- [email protected] (CP) tion of biliary and enteral drainage were performed. A fro- Copyright: © 2021 The Author(s). This article has been published under the terms of Creative Commons Attribution-Noncommercial 4.0 International License (CC BY-NC 4.0), which permits noncommercial unrestricted use, distribution, and reproduction in any medium, provided that the following statement is provided. “This article has been published in Journal of Clinical and Translational Hepatology at https://doi.org/10.14218/JCTH.2020.00123 and can also be viewed on the Journal’s website at http://www.jcthnet.com ”. Liu S. et al: Mucinous cystic tumor of gallbladder Fig. 1. Preoperative imaging revealed biliary obstruction. (A, B) Abdominal plain computed tomography scan plus enhanced (C) Magnetic resonance choliangio- pancreatography 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. zen section was assessed intraoperatively, and the results Board of Hunan Provincial People’s Hospital/The First Affili- were reported as: “Consider gallbladder cyst adenoma”. We ated Hospital of Hunan Normal University. then performed an open cholecystectomy, biliary explora- tion, and bile duct repair with shaping and T-tube drainage. During the operation, a 4.0×2.0 cm mass of the cystic Discussion duct protruded into the bile duct lumen (Fig. 2A–D). The mass grew on the wall of the cystic duct with a pedicle, and Mucinous cystic neoplasms (MCNs) were first reported in a few stones were seen in the gallbladder. pancreatic tissue and, subsequently, there has been much The predischarge inspection showed that the total biliru- research devoted to investigating pancreatic MCNs. Howev- bin was 25.8 μmol/L and the direct bilirubin was 18 μmol/L. er, there are still many controversies about pancreatic MCN Computed tomography of the abdomen showed that the disease and even less is known about gallbladder MCN. Ac- dilatation of the bile ducts, inside and outside the liver, cording to the authors’ search of the PubMed database, the was significantly less than before. Postoperative pathology earliest case of gallbladder MCN was reported by Bishop showed that there was a multicystic mass in the cystic duct, in The Lancet in 1901,1 and there have been 16 literature 4×2×2 cm in size, multicystic at the cut surface, and con- reports on gallbladder MCN (Table 1). taining light-yellow, clear liquid in the cyst. The pathological Similar to pancreatic MCN, gallbladder MCN can manifest diagnosis was mucinous cystic tumor with mild atypical hy- unilocular or multilocular cystic changes, containing septa. perplasia with chronic cholecystitis immunohistochemistry In the World Health Organization Classification of Digestive of cytokeratin 7 (+), cytokeratin 19 (epithelial +), estrogen System Tumors (2010 Edition), biliary MCN is listed sepa- receptor (+), progesterone receptor (+), P53 (−), and Ki67 rately, as a special tumor of the gallbladder, and is classified (scattered +) (Fig. 3). into “mucocystic tumors with low-grade or medium-grade epithelium according to the status of intraepithelial neopla- Informed consent sia. Internal neoplasia (8470/0) (8470/2)”, “Invasive mu- cocystic carcinoma (8470/3)”. The existing literature data divides MCN into at least two types.1 One is non-invasive Prior written informed consent was provided from the pa- and has ovarian-like stroma under the epithelium, which is tient and this study was approved by the Ethics Review characterized by a high cell density. It appears as a dense 2 Journal of Clinical and Translational Hepatology 2021 vol. 000 | 000–000 Liu S. et al: Mucinous cystic tumor of gallbladder 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. collection of spindle-shaped cells lacking cytoplasm and is 95% for adenoma and 63% for cancer, among which micro- immune to estrogen and progesterone receptors. This sub- invasive carcinoma also reached more than 90%.4 Another type affects middle-aged women. The other type is more study showed that the 5-year survival rate of untreated pan- aggressive, has no ovarian-like stroma, and affects men creatic MCN with invasive carcinoma was about 30% and between 75 and 88 years-old. There are others who clas- the prognosis was poor.5 Such statistics are still lacking for sify MCN using three subtypes, based on epithelial atypia gallbladder MCN.
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