Abstract
BackgroundAn outbreak of cholangiocarcinoma in Japan has led to widespread concern among workers in printing plants. In March 2013, the Japanese Ministry of Health, Labour and Welfare, confirmed a causal relationship between cholangiocarcinoma and long-term exposure to dichloromethane (DCM) and 1,2-dichloropropane (DCP), which were widely used in printing plants. We herein report a rare case of successful radical resection of multiple cholangiocarcinomas in the intrahepatic and extrahepatic bile ducts caused by past exposure to DCM.Case presentationA 54-year-old man developed brown urine 22 years after his last exposure to DCP and DCM. He had an 11-year history of working at a printing plant from the age of 21 to 31 years and dealt with organic solvents during his employment. Enhanced computed tomography revealed a thickened distal bile duct wall with upstream biliary dilatation and multiple intrahepatic cholangiocarcinomas located in liver segments III, VI, and VIII. Biopsy of the distal bile duct wall revealed adenocarcinoma, and a diagnosis of distal cholangiocarcinoma was made. Tumor marker levels were within the reference range (carcinoembryonic antigen, 3.3 ng/mL; carbohydrate antigen 19-9, 25.4 U/mL; SPAN-1, 13 U/mL; and DUPAN-2, 33 U/mL). The multiple intrahepatic and extrahepatic bile duct cancers were treated by subtotal stomach-preserving pancreatoduodenectomy and partial hepatectomy of segments III, VI, and VIII. Pathological examination of the surgical specimens revealed multiple cholangiocarcinomas with well-differentiated adenocarcinoma in the biliary tree. The patient was still alive without recurrence 17 months after the operation.ConclusionsWe experienced a rare case of multiple cholangiocarcinomas in the intrahepatic and extrahepatic bile ducts that developed 22 years after the patient’s last exposure to DCP and DCM. Long-term and careful follow-up is required for workers with an occupational history of exposure to organic solvents because of the risk of development of cholangiocarcinoma.
Highlights
An outbreak of cholangiocarcinoma in Japan has led to widespread concern among workers in printing plants
We experienced a rare case of multiple cholangiocarcinomas in the intrahepatic and extrahepatic bile ducts that developed 22 years after the patient’s last exposure to DCP and DCM
A previous study reported that only two cases of multiple intrahepatic and extrahepatic occupational cholangiocarcinoma with radical resection have been described
Summary
The characteristics of occupational cholangiocarcinoma include elevated serum gamma-glutamyl transferase activity, regional dilatation of the bile duct without tumor-induced obstruction and biliary findings similar to those of primary sclerosing cholangitis on diagnostic imaging, the presence of precancerous or early cancerous lesions, and nonspecific bile duct injuries [1, 3, 5, 10,11,12]. In the present case, the radical resection for multiple cholangiocarcinomas in the intrahepatic and extrahepatic biliary tree could provide a favorable prognostic outcome as in the previous reports [1, 11]. In this case, it is difficult to exclude the possibility of intrahepatic metastasis from extrahepatic biliary duct cancer. Long-term and careful follow-up is required for workers with an occupational history of exposure to high concentrations of DCM and/ or DCP because of the possibility of the development of cholangiocarcinoma. To ensure an accurate diagnosis of occupational cholangiocarcinoma, laboratory data and radiological findings and a confirmed history of working at a printing plant is very important
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