Ann Hepatobiliary Pancreat Surg.  2020 May;24(2):209-215. 10.14701/ahbps.2020.24.2.209.

Living donor liver transplantation for diffuse biliary papillomatosis with malignant change: A case report with 10-year follow-up

• Choi JU
• Hwang S
• Jung DH
• Park GC
• Ahn CS
• Kim KH
• Moon DB
• Ha TY
• Song GW
• Lee SG

• KMID: 2500808
• DOI: http://doi.org/10.14701/ahbps.2020.24.2.209

Abstract

Biliary papillomatosis is a rare disease of the biliary tract. In this study, we present a case of a male patient with biliary papillomatosis with malignant change who underwent living donor liver transplantation (LDLT). The 49-year-old man was admitted due to intrahepatic ductal mass, which was finally diagnosed as biliary papillomatosis. It was initially treated by photodynamic therapy, but LDLT was planned due to low efficacy of photodynamic therapy. LDLT was performed with a right liver graft from his son. The pathology report revealed intraductal papillary neoplasm with associated invasive carcinoma. He recovered uneventfully. Although the distal bile duct resection margin was free of tumor, the risk of tumor recurrence at the remnant intrahepatic bile duct still remained. Therefore, staged pylorus-preserving pancreatoduodenectomy was planned. However, after 3 months, he was readmitted due to retropancreatic abscess. It took 6 months to control the intractable retropancreatic abscess. Due to late development of retropancreatic abscess and its slow recovery, staged pancreatoduodenectomy was no longer considered. The patient recovered complete at 1 year posttransplant without any evidence of disease recurrence. After 5 years posttransplant, lung metastasis developed and pulmonary metastasectomy was performed three times. No further recurrence developed during the last 3 years to date, and the patient currently is alive, 10 years after the LDLT. Experience on our case and literature review suggest that patients with biliary papillomatosis with or without malignant transformation are selectively indicated for liver transplantation.

Keyword

Biliary papillomatosis; Intraductal papillary neoplasm; Living donor liver transplantation; Lung metastasis

Figure

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  Fig. 1 Pretransplant imaging study findings. Dynamic computed tomography scan images (A and B) and magnetic resonance pancreatocholangiography (C) showed diffuse intraductal papillomatosis.

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  Fig. 2 Cholangiography findings. A percutaneous transhepatic biliary drainage catheter was inserted (A) and percutaneous transhepatic cholangioscopy was performed (B).

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  Fig. 3 Cholangioscopic findings. (A) The lumens of the whole intrahepatic ducts were covered with villotubular adenomas with focal high-grade dysplasia. (B) There were areas showing atypical villous proliferation, which was consistent with well-differentiated adenocarcinoma.

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  Fig. 4 Photodynamic therapy. Transcatheter photodynamic therapy was performed (A) to the intrahepatic biliary papillomatosis areas (B-D).

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  Fig. 5 Dynamic computed tomography scan taken 2 weeks after living donor liver transplantation. It was a typical posttransplant finding using a modified right liver graft.

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  Fig. 6 Gross photographs of the explant liver. (A) No morphological change of the external contour of the liver was observed. (B) The intrahepatic ducts were lined with papillomatosis and the common bile duct was discolored by damage from precedent photodynamic therapy.

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  Fig. 7 Serial follow-up with computed tomography (CT) scans for retropancreatic abscess. (A) Retropancreatic abscess was first detected at the 3-month CT scan. (B) The 5-month CT scan showed slightly improvement with pigtail drainage. (C) The 7-month CT scan showed persistence of retropancreatic abscess.

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  Fig. 8 Dynamic computed tomography scan taken 1 year after living donor liver transplantation. There was no abnormality at the liver graft (A) and retropancreatic abscess was completely resolved (B).

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  Fig. 9 Serial follow-up with computed tomography (CT) scans for lung metastasis. (A) A metastatic nodule was detected after 5 years (arrow). (B) Another metastatic nodule was detected after 6 years (arrow). (C) The other metastatic nodule was detected after 7 years (arrow). (D) Gross photograph showed the excised metastatic nodule shown at the 7-year CT scan.

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