Ann Liver Transplant.  2022 May;2(1):86-94. 10.52604/alt.22.0001.

Development of perihilar cholangiocarcinoma at 29 years after first hepatectomy for hepatolithiasis

• Kim SM
• Hwang S
• Kim Y
• Jwa EK

• KMID: 2560886
• DOI: http://doi.org/10.52604/alt.22.0001

Abstract

Hepatolithiasis is a known risk factor for intrahepatic cholangiocarcinoma. We present a case of a patient with perihilar cholangiocarcinoma that arose from the remnant hilar bile duct at 29 years after the initial hepatectomy for left-sided hepatolithiasis and at 16 years after completion left hepatectomy. A 66-year-old female patient was diagnosed with 4 cm-sized perihilar cholangiocarcinoma at follow-up studies. The tumor appeared to be confined at the right first-order hepatic duct without gross vascular invasion, thus surgical resection was performed with a curative intent. The tumor-containing right first-order hepatic duct was meticulously resected with concurrent removal of the caudate lobe. Cluster hepaticojejunostomy was performed to reconstruct the 4 cm-wide figure of 8-shaped intrahepatic bile duct openings. Significant bile leak occurred at the hepaticojejunostomy site, which was resolved with percutaneous pigtail drainage and percutaneous transhepatic biliary drainage. The patient was discharged from the hospital at 26 days after operation. The patient has been doing well for 12 months. She is currently undergoing scheduled adjuvant chemotherapy. The experience of our present case suggests that there is risk of de novo hepatobiliary malignancy after hepatic resection for hepatolithiasis. Thus, it is necessary to perform life-long follow-up studies for patients who have undergone hepatic resection for hepatolithiasis.

Keyword

Malignant transformation; Hepatolithiasis; Cholangiocarcinoma; Hepatic resection; Long-term follow-up

Figure

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  Figure 1 Prerioperative imaging study findings before the completion left hepatectomy. (A) Preoperative computed tomography (CT) shows markedly atrophy of the remnant left medial section filled with multiple intrahepatic stones. (B, C) Preoperative magnetic resonance cholangiography shows multiple intrahepatic stones in the remnant left medial section and right lobe. (D) Post-hepatectomy CT shows complete removal of the hepatolithiasis-involved the remnant left medial section.

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  Figure 2 Management of recurrent hepatolithiasis at 4 years after completion left hepatectomy. (A) Magnetic resonance cholangiography shows intrahepatic duct stenosis with stone formation at segment VI. (B, C) Segmental stenosis at the right posterior hepatic duct was dilated and intrahepatic duct stones were removed through percutaneous transhepatic cholangioscopy. (D) Follow-up computed tomography shows resolution of intrahepatic duct stenosis and loss of intrahepatic duct stones.

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  Figure 3 Preoperative computed tomography (CT) findings taken at 16 years after the completion left hepatectomy. (A) A 3-cm-sized intraductal mass (arrow) is identified at the first-order right hepatic duct and abscess is formed at the caudate lobe. (B, C) Follow-up CT taken 1 month later shows marked growth of the intraductal tumor (arrows). (D) The right hepatic artery is not encased with the tumor.

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  Figure 4 Preoperative cholangiography findings taken at 16 years after the completion left hepatectomy. (A, B) Magnetic resonance cholangiography shows intraductal mass (arrow) with marked dilatation of the right hepatic duct. (C, D) Endoscopic retrograde cholangiography is performed for tissue biopsy and two endoscopic retrograde biliary drainage tubes are inserted.

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  Figure 5 Preoperative fluorodeoxyglucose positron emission tomography finding. A mass of hypermetabolic uptake is visible at the hepatic hilum with mild hypermetabolic lymph node enlargement at the aortocaval area.

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  Figure 6 Intraoperative photographs showing resection of the tumor. (A) The choledochoduodenostomy (arrow) was transected to make the operative field wide. (B) The common bile duct, the right hepatic artery and the main portal vein are isolated with transection of the distal bile duct. Arrow indicated the site of choledochoduodenostomy. (C) The enlarged first-order right hepatic duct is dissected from the right portal vein and the right hepatic artery. (D) The first-order right hepatic duct (arrow) is transected after palpation of the intraductal mass. (E, F) The right anterior and posterior intrahepatic bile duct openings (arrows) are exposed as a figure of 8.

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  Figure 7 Intraoperative photographs showing the procedures of cluster hepaticojejunostomy. (A) The anterior wall of the bile duct openings is anchored with multiple 5-0 Prolene sutures. (B, C) The posterior wall of the bile duct openings is continuously sutured with 5-0 Prolene sutures after dividing it into four segments with three internal intervening sutures. (D) The anterior wall is closed by using interrupted sutures that are previously anchored.

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  Figure 8 Gross photograph of the resected specimen showing an intraductal mass.

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  Figure 9 Postoperative computed tomography findings taken at 3 days after the second operation. (A, B) Cross-sectional images show the extent of resection. (C, D) The reconstructed second-order right hepatic duct shows good patency.

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  Figure 10 Fig. 10 . Postoperative computed tomography findings taken 9 months after the third second hepatectomy with bile duct resection. The cross-sectional image (A) and reconstructed image (B) show no abnormal postoperative findings.

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