A case of small in situ perihilar cholangiocarcinoma incidentally accompanied by benign bile duct stricture after open cholecystectomy
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In situ cholangiocarcinoma is difficult to detect by imaging studies. Thus, cholangiocarcinoma is rarely resected with a preoperative definitive diagnosis, especially nonpapillary flat type in situ carcinoma, which is extremely rare.
A 70-year old man was diagnosed with gallbladder cancer and received open cholecystectomy with lymphadenectomy at a local hospital. Histologically, the tumor was localized in the mucosal layer, and no lymph node metastases were found. Three months later, hilar bile duct stricture due to delayed bile duct ischemia was found. Then, biliary drainage was performed with endoscopic biliary stenting. Three months later, the patient experienced cholangitis with septic shock, and percutaneous transhepatic biliary drainage (PTBD) into the left intrahepatic bile duct was performed. Unexpectedly, the aspiration bile cytology of the PTBD catheter showed malignant cells, and the patient was referred to our clinic for possible surgical treatment. According to additional studies, the hilar bile duct stricture was 3 cm in length. None of the imaging studies detected malignant cells in the bile duct around the hilar stricture. The left portal vein was obstructed due to inadvertent puncture of the PTBD. No findings indicated cholangiocarcinoma. We performed left hepatectomy with caudate lobectomy and extrahepatic bile duct resection. The postoperative course was uneventful. In the final pathology, flat type in situ carcinoma was found at the confluence of the right and left hepatic ducts, which was distant from the biliary stricture.
When a tumor is undetectable but cytology is positive, in situ cholangiocarcinoma may exist; thus, surgery should be carefully considered.
KeywordsBenign bile duct stricture Carcinoma in situ Perihilar cholangiocarcinoma Aspiration bile cytology
Common bile duct
Endoscopic nasobiliary drainage
Multidetector-row computed tomography
Percutaneous transhepatic biliary drainage
Percutaneous transhepatic cholangioscopy
In situ cholangiocarcinoma, i.e., epithelial carcinoma without submucosal invasion, is asymptomatic and very difficult to detect by multidetector-row computed tomography (MDCT) or direct cholangiography. Thus, this type of carcinoma is rarely resected with a preoperative definitive diagnosis, especially, nonpapillary flat type in situ carcinoma, which is extremely rare.
Here, we present a case of flat type in situ perihilar cholangiocarcinoma that was incidentally accompanied by hilar bile duct stricture after open cholecystectomy. Preoperative diagnostic imaging studies did not identify the lesion, and only aspiration bile cytology showed a positive result.
When arterial blood flow to the bile duct is restricted, a bile duct stricture will develop [1, 2]. Several authors have reported delayed bile duct strictures due to ischemia after excision of the tissue surrounding the biliary tree [3, 4, 5]. Ishizuka et al.  reported two cases of delayed ischemic biliary stricture after radical lymphadenectomy in the hepatoduodenal ligament with skeletonization of the extrahepatic bile ducts for malignant diseases: in both cases, histologic examination of the subsequently resected biliary strictures revealed evidence of ischemia. Skeletonization of the extrahepatic bile duct may induce ischemia then delayed stricture formation. Lymphadenectomy of the hepatoduodenal ligament is routinely performed in advanced gallbladder carcinoma. However, when preserving the extrahepatic bile duct, this surgical procedure may induce bile duct stricture, and the present case is a case of delayed ischemic stricture. For prevention of this complication, assessment of arterial perfusion in the bile duct wall using indocyanine green near-infrared imaging may be a promising way .
Flat type precursor lesions are called biliary intraepithelial neoplasias and are classified into three grades . Especially with severe atypia, the lesions are identical to in situ carcinoma. This kind of epithelial carcinoma is often detected as a lesion accompanied by invasive carcinoma , while in situ carcinoma alone is rarely detected. According to our previous study , only 3 cases of in situ carcinoma were found in 545 consecutive resections of perihilar cholangiocarcinoma. Thus, the incidence of in situ perihilar cholangiocarcinoma was 0.55%.
In this case, aspiration bile cytology alone showed a positive result. We performed aspiration cytology from the PTBD catheter five times, which was positive three times. Tsuchiya et al.  reported that the positivity of aspiration bile cytology increased when repeatedly performed but reached a plateau after five examinations. Aspiration cytology is easy to perform and repeatable ; thus, it should be used, especially in cases of negative biopsy results. However, aspiration cytology never indicates location of the lesion, which is a major limitation of this examination.
We experienced a rare case of flat type in situ perihilar cholangiocarcinoma that was incidentally accompanied by a benign bile duct stricture. When the tumor is undetectable but aspiration cytology is positive, in situ cholangiocarcinoma may exist; thus, surgery should be carefully considered.
We would like to thank American Journal Experts (www.aje.com) for the English language editing.
MN, NW, MN, and KS performed the surgery and perioperative management on the patient, and MN drafted the manuscript. All authors read and approved the final manuscript.
The authors declare that they received no financial support pertaining to this case report.
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Written informed consent was obtained from the patient for the publication of this case report and any accompanying images.
The authors declare that they have no competing interests.
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