Ex) Article Title, Author, Keywords
Ex) Article Title, Author, Keywords
Ann Liver Transplant 2023; 3(1): 57-62
Published online May 31, 2023 https://doi.org/10.52604/alt.23.0008
Copyright © The Korean Liver Transplantation Society.
Dae Hyeon Won , Shin Hwang
, Yumi Kim
Correspondence to:Shin Hwang
Department of Surgery, Asan Medical Center, University of Ulsan College of Medicine, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 05505, Korea
E-mail: shwang@amc.seoul.kr
https://orcid.org/0000-0002-9045-2531
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/bync/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Choledochal cyst is associated with biliary malignancies such as bile duct cancer and gallbladder cancer. We herein present a case of preemptive choledochal cyst resection during redo operation for incidentally detected gallbladder cancer following laparoscopic cholecystectomy. A 64-year-old female patient underwent laparoscopic cholecystectomy and gallbladder cancer was diagnosed. Imaging studies revealed that the patient had choledochal cyst with anomalous union of the pancreaticobiliary duct. Preoperative findings suggested that gallbladder cancer might have be developed due to the carcinogenic effect of the anomalous union of the pancreaticobiliary duct-associated pancreatic reflux. Thus, preemptive resection of choledochal cyst was performed with redo radical cholecystectomy. The patient recovered uneventfully. The tumor stage was T2N0M0, tumor-node-metastasis stage II. The patient has been undergoing adjuvant chemotherapy. The present case suggests that preemptive bile duct resection can be considered in gallbladder cancer patients with anomalous union of the pancreaticobiliary duct and asymptomatic choledochal cyst.
Keywords: Gallbladder cancer, Bile duct cancer, Choledochal cyst, Malignant transformation, Anomalous union of pancreaticobiliary duct
Choledochal cyst disease (CCD) is a pathologic condition that is characterized by varying degrees of congenital dilatation of the biliary system, including the common, intrahepatic, and intrapancreatic bile ducts. CCD is not uncommon in Asian patients, especially in females and infants [1-8]. The diagnosis of CCD in adults has become more frequent over time than previously [7,8]. Many of these patients may have long-standing CCD, but might not be diagnosed until adulthood [8]. CCD is associated with biliary malignancies such as bile duct cancer and gallbladder cancer [9,10]. The pathogenesis of cancer in CCDs may be associated with the carcinogenic effect of pancreatic reflux. The risk of cholangiocarcinoma in cysts is much higher in patients with anomalous union of the pancreaticobiliary duct (AUPBD) [11]. We herein present a case of preemptive CCD resection during redo operation for incidentally detected gallbladder cancer following laparoscopic cholecystectomy.
A 64-year-old female patient was transferred for the treatment of gallbladder cancer that had been diagnosed after laparoscopic cholecystectomy. Pathology finding of the gallbladder was poorly differentiated adenocarcinoma with focal signet ring cell carcinoma component, and tumor depth was muscle layer invasion (pT2). The patient had no history of specific hepatobiliary disease before. Serum cancer antigen 19-9 was not elevated.
Review of the preoperative abdomen computed tomography scans revealed gallbladder wall thickening had occurred at the background of the CCD (Fig. 1–3). Magnetic resonance cholangiopancreatography showed the presence of AUPBD (Fig. 4). These findings suggested that gallbladder cancer might have developed due to the carcinogenic effect of AUPBD-associated pancreatic reflux. Thus, preemptive resection of CCD might be reasonably indicated.
At 4 weeks after laparoscopic cholecystectomy, redo surgery was performed. Heavy adhesion of the greater omentum at the gallbladder bed was meticulously removed through omentectomy. Dissection of the hepatoduodenal ligament revealed definite fusiform bulging of the common bile duct (CBD), indicating CCD. The perihilar structures were also heavily adhered not permitting sharp dissection, thus en bloc hepatic resection of the gallbladder bed was performed. Thereafter, the distal end of the CCD was transected at 5 mm above the level of the intrapancreatic CBD narrowing to prevent iatrogenic pancreatic duct injury. Distal CBD resection margin was tumor-negative on intraoperative frozen-section biopsy. This cephalad dissection of the CCD facilitated sharp dissection of the hepatoduodenal ligament. The CCD wall was longitudinally incised to identify the location of hilar bifurcation. The proximal part of the CCD was transected at 5 mm distal to hilar bifurcation to facilitate biliary reconstruction. Proximal CBD resection margin was tumor-negative on intraoperative frozen-section biopsy. An en block mass of the liver bed and CCD was removed. Extensive lymph node dissection was performed along the retropancreatic, common hepatic artery, and celiac axis areas (Fig. 5). Roux-en-Y hepaticojejunostomy was performed with combination of posterior running sutures and anterior interrupted sutures.
The pathology report revealed no residual tumor and no lymph node metastasis, indicating pT2N0M0, tumor-node-metastasis stage II (Fig. 6). The patient recovered uneventfully from this extended cholecystectomy with bile duct resection (Fig. 7). Considering this tumor stage, the patient has been undergoing adjuvant chemotherapy.
The debatable issue in the present case is whether concurrent resection of the CCD is reasonably indicated for a middle-aged patient of over 60 years age. The present case had typical type I CCD with AUPBD, and the development of gallbladder cancer might be highly associated with the carcinogenic effect of AUPBD-associated pancreatic reflux. To prevent late malignant transformation in the CCD, we decided to perform concurrent resection of the CCD.
A considerable proportion of CCD patients had concomitant cancer. In a Japanese registry of 1,353 patients with CCD and/or AUPBD, 16% had coincident cancer, increasing with each decade of life, from 2% for patients in their 20s to 43% for those in their 60s [12]. When present, cancer occurs in patients at least one decade younger than in typical patients with gallbladder cancer or cholangiocarcinoma [8]. We reported concurrent cancers in 9.8% of CCD patients and the median ages of patients with bile duct and gallbladder cancers were 45 years and 52 years, respectively [9]. A Korean single-center study revealed that 10 of 218 patients (4.6%) with gallbladder cancers were associated with AUPBD and seven of them (70%) had concurrent CCD [10].
The malignant transformation in CCDs may be associated with the carcinogenic effect of pancreatic reflux. The risk of cholangiocarcinoma in CCDs is much higher in patients with AUPBD (32%) than without AUPBD (0%) [11]. We previously reported that AUPBD was more closely associated with the incidence of gallbladder cancer than bile duct cancer [9].
Whenever possible, CCD should be completely resected, due to the long-term consequences of cholangitis, liver cirrhosis, pancreatitis, and malignant transformation [13]. Although all portions of the CCDs should be removed, residual proximal cyst walls may be left to facilitate biliary anastomosis [14,15]. Complete cyst excision requires accurate recognition of the origin and termination of the cyst. The ends of cysts extending from the confluence of the hepatic duct to the junction of the common duct and pancreatic duct are very difficult to clearly define. It is also difficult to differentiate the normal bile duct endothelium from the cyst lining by using intraoperative frozen-section biopsies. Our procedure includes opening the cyst wall and and grossly identifying the luminal appearance to determine the proximal transection line. This effectively reduces the risks of intractable anastomotic stricture.
During dissection of the intrapancreatic cyst, there is a potential risk of pancreatic duct injury. Several methods have been proposed to remove the intrapancreatic cyst without pancreatic duct injury, including intraoperative endoscopy and intraoperative cholangiography after the placement of hemoclips [16,17]. We have utilized preoperative imaging to decide the extent of intrapancreatic dissection in combination with open inspection and probing of the intrapancreatic distal bile duct.
The present case suggests that preemptive bile duct resection can be considered in gallbladder cancer patients with AUPBD and asymptomatic CCD.
There was no funding related to this study.
All authors have no conflicts of interest to declare.
Conceptualization: SH. Data curation: DHW. Formal analysis: All. Methodology: All. Writing - original draft: All. Writing - review & editing: DHW, SH.
Ann Liver Transplant 2023; 3(1): 57-62
Published online May 31, 2023 https://doi.org/10.52604/alt.23.0008
Copyright © The Korean Liver Transplantation Society.
Dae Hyeon Won , Shin Hwang
, Yumi Kim
Department of Surgery, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
Correspondence to:Shin Hwang
Department of Surgery, Asan Medical Center, University of Ulsan College of Medicine, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 05505, Korea
E-mail: shwang@amc.seoul.kr
https://orcid.org/0000-0002-9045-2531
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/bync/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Choledochal cyst is associated with biliary malignancies such as bile duct cancer and gallbladder cancer. We herein present a case of preemptive choledochal cyst resection during redo operation for incidentally detected gallbladder cancer following laparoscopic cholecystectomy. A 64-year-old female patient underwent laparoscopic cholecystectomy and gallbladder cancer was diagnosed. Imaging studies revealed that the patient had choledochal cyst with anomalous union of the pancreaticobiliary duct. Preoperative findings suggested that gallbladder cancer might have be developed due to the carcinogenic effect of the anomalous union of the pancreaticobiliary duct-associated pancreatic reflux. Thus, preemptive resection of choledochal cyst was performed with redo radical cholecystectomy. The patient recovered uneventfully. The tumor stage was T2N0M0, tumor-node-metastasis stage II. The patient has been undergoing adjuvant chemotherapy. The present case suggests that preemptive bile duct resection can be considered in gallbladder cancer patients with anomalous union of the pancreaticobiliary duct and asymptomatic choledochal cyst.
Keywords: Gallbladder cancer, Bile duct cancer, Choledochal cyst, Malignant transformation, Anomalous union of pancreaticobiliary duct
Choledochal cyst disease (CCD) is a pathologic condition that is characterized by varying degrees of congenital dilatation of the biliary system, including the common, intrahepatic, and intrapancreatic bile ducts. CCD is not uncommon in Asian patients, especially in females and infants [1-8]. The diagnosis of CCD in adults has become more frequent over time than previously [7,8]. Many of these patients may have long-standing CCD, but might not be diagnosed until adulthood [8]. CCD is associated with biliary malignancies such as bile duct cancer and gallbladder cancer [9,10]. The pathogenesis of cancer in CCDs may be associated with the carcinogenic effect of pancreatic reflux. The risk of cholangiocarcinoma in cysts is much higher in patients with anomalous union of the pancreaticobiliary duct (AUPBD) [11]. We herein present a case of preemptive CCD resection during redo operation for incidentally detected gallbladder cancer following laparoscopic cholecystectomy.
A 64-year-old female patient was transferred for the treatment of gallbladder cancer that had been diagnosed after laparoscopic cholecystectomy. Pathology finding of the gallbladder was poorly differentiated adenocarcinoma with focal signet ring cell carcinoma component, and tumor depth was muscle layer invasion (pT2). The patient had no history of specific hepatobiliary disease before. Serum cancer antigen 19-9 was not elevated.
Review of the preoperative abdomen computed tomography scans revealed gallbladder wall thickening had occurred at the background of the CCD (Fig. 1–3). Magnetic resonance cholangiopancreatography showed the presence of AUPBD (Fig. 4). These findings suggested that gallbladder cancer might have developed due to the carcinogenic effect of AUPBD-associated pancreatic reflux. Thus, preemptive resection of CCD might be reasonably indicated.
At 4 weeks after laparoscopic cholecystectomy, redo surgery was performed. Heavy adhesion of the greater omentum at the gallbladder bed was meticulously removed through omentectomy. Dissection of the hepatoduodenal ligament revealed definite fusiform bulging of the common bile duct (CBD), indicating CCD. The perihilar structures were also heavily adhered not permitting sharp dissection, thus en bloc hepatic resection of the gallbladder bed was performed. Thereafter, the distal end of the CCD was transected at 5 mm above the level of the intrapancreatic CBD narrowing to prevent iatrogenic pancreatic duct injury. Distal CBD resection margin was tumor-negative on intraoperative frozen-section biopsy. This cephalad dissection of the CCD facilitated sharp dissection of the hepatoduodenal ligament. The CCD wall was longitudinally incised to identify the location of hilar bifurcation. The proximal part of the CCD was transected at 5 mm distal to hilar bifurcation to facilitate biliary reconstruction. Proximal CBD resection margin was tumor-negative on intraoperative frozen-section biopsy. An en block mass of the liver bed and CCD was removed. Extensive lymph node dissection was performed along the retropancreatic, common hepatic artery, and celiac axis areas (Fig. 5). Roux-en-Y hepaticojejunostomy was performed with combination of posterior running sutures and anterior interrupted sutures.
The pathology report revealed no residual tumor and no lymph node metastasis, indicating pT2N0M0, tumor-node-metastasis stage II (Fig. 6). The patient recovered uneventfully from this extended cholecystectomy with bile duct resection (Fig. 7). Considering this tumor stage, the patient has been undergoing adjuvant chemotherapy.
The debatable issue in the present case is whether concurrent resection of the CCD is reasonably indicated for a middle-aged patient of over 60 years age. The present case had typical type I CCD with AUPBD, and the development of gallbladder cancer might be highly associated with the carcinogenic effect of AUPBD-associated pancreatic reflux. To prevent late malignant transformation in the CCD, we decided to perform concurrent resection of the CCD.
A considerable proportion of CCD patients had concomitant cancer. In a Japanese registry of 1,353 patients with CCD and/or AUPBD, 16% had coincident cancer, increasing with each decade of life, from 2% for patients in their 20s to 43% for those in their 60s [12]. When present, cancer occurs in patients at least one decade younger than in typical patients with gallbladder cancer or cholangiocarcinoma [8]. We reported concurrent cancers in 9.8% of CCD patients and the median ages of patients with bile duct and gallbladder cancers were 45 years and 52 years, respectively [9]. A Korean single-center study revealed that 10 of 218 patients (4.6%) with gallbladder cancers were associated with AUPBD and seven of them (70%) had concurrent CCD [10].
The malignant transformation in CCDs may be associated with the carcinogenic effect of pancreatic reflux. The risk of cholangiocarcinoma in CCDs is much higher in patients with AUPBD (32%) than without AUPBD (0%) [11]. We previously reported that AUPBD was more closely associated with the incidence of gallbladder cancer than bile duct cancer [9].
Whenever possible, CCD should be completely resected, due to the long-term consequences of cholangitis, liver cirrhosis, pancreatitis, and malignant transformation [13]. Although all portions of the CCDs should be removed, residual proximal cyst walls may be left to facilitate biliary anastomosis [14,15]. Complete cyst excision requires accurate recognition of the origin and termination of the cyst. The ends of cysts extending from the confluence of the hepatic duct to the junction of the common duct and pancreatic duct are very difficult to clearly define. It is also difficult to differentiate the normal bile duct endothelium from the cyst lining by using intraoperative frozen-section biopsies. Our procedure includes opening the cyst wall and and grossly identifying the luminal appearance to determine the proximal transection line. This effectively reduces the risks of intractable anastomotic stricture.
During dissection of the intrapancreatic cyst, there is a potential risk of pancreatic duct injury. Several methods have been proposed to remove the intrapancreatic cyst without pancreatic duct injury, including intraoperative endoscopy and intraoperative cholangiography after the placement of hemoclips [16,17]. We have utilized preoperative imaging to decide the extent of intrapancreatic dissection in combination with open inspection and probing of the intrapancreatic distal bile duct.
The present case suggests that preemptive bile duct resection can be considered in gallbladder cancer patients with AUPBD and asymptomatic CCD.
There was no funding related to this study.
All authors have no conflicts of interest to declare.
Conceptualization: SH. Data curation: DHW. Formal analysis: All. Methodology: All. Writing - original draft: All. Writing - review & editing: DHW, SH.