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Portal Vein Tumor Thrombus Originating from Ampullary Adenocarcinoma: A Case Report

Portal Vein Tumor Thrombus Originating from Ampullary Adenocarcinoma: A Case Report Journal of Pancreatology DOI: 10.1097/JP9.0000000000000226 Portal Vein Tumor Thrombus Originating from Ampullary Adenocarcinoma: A Case Report Ye Cao, Qiyuan Li, Binru Zhang, Yongwei Wang, Bei Sun, Hua Chen Department of Pancreatic and Biliary Surgery, The First Affiliated Hospital of Harbin Medical University, Harbin, China Corresponding Author: Hua Chen (Email:[email protected]) Authors’ contributions Ye Cao, Qiyuan Li, Binru Zhang wrote the manuscript. Yongwei Wang, Bei Sun, Hua Chen performed the surgery. All authors conceived the study, participated in its design and coordination, and helped to draft the manuscript. All authors have read and approved the final manuscript. Consent for publication Written informed consent was obtained from the patient for publication of this case report and any accompanying images. Ethical Approval This case report represents a fully anonymized retrospective presentation. Therefore, the need for ethical approval was waived according to the regulation of the Ethics Committee of The First Affiliated Hospital of Harbin Medical University. This case report was not registered in any online public registration platform. ACCEPTED Competing interests The authors declare that they have no competing interests. This is an open access article distributed under the terms of the Creative Commons Attribution- Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. ACCEPTED Abstract: We present a rare case of a 62-year-old female with ampullary adenocarcinoma complicated by portal vein tumor thrombus (PVTT). The patient presented with upper abdominal pain, jaundice, and fever. Imaging revealed a distal common bile duct mass with portal vein thrombosis. After multidisciplinary discussion, she underwent laparoscopic pancreaticoduodenectomy with portal vein resection and reconstruction. Histopathology confirmed the presence of adenocarcinoma and PVTT. The patient had an uneventful recovery and is disease-free at 6 months post-surgery. Keywords: Ampullary carcinoma; Portal vein tumor thrombus; Metastases ACCEPTED 1. Introduction Ampullary carcinoma is a rare malignant tumor of the gastrointestinal tract, classified as a type of peri-ampullary cancer. Common metastatic sites include the liver, peritoneum, and lungs. [1] Additionally, there are reports of ampullary carcinoma metastasizing to the bones, brain , and [2] orbit . Portal vein tumor thrombosis (PVTT) in ampullary carcinoma is exceedingly rare, and a systematic literature search conducted across PubMed, EMBASE, and Web of Science databases confirmed that no cases of this specific tumor entity have been reported in the English-language medical literature to date. We present the first documented case of PVTT associated with ampullary carcinoma. 2. Case Report A 62-year-old female presented with a one-week history of upper abdominal pain accompanied by fever, jaundice, nausea, and abdominal distension. Six months prior, she had undergone laparoscopic common bile duct exploration for gallbladder and bile duct stones. Admission laboratory tests revealed: ALT: 115 U/L, AST: 122 U/L, TBIL: 107.6 μmol/L, DBIL: 87.9 μmol/L, and CA19-9: 99.71 U/mL. Magnetic resonance cholangiopancreatography (MRCP) demonstrated a distal common bile duct stone with secondary intrahepatic and extrahepatic bile duct dilation. Contrast-enhanced pancreatic CT revealed a mass in the distal common bile duct causing low bile duct obstruction and thrombosis in the main portal vein (Figure 1A-B). To confirm the diagnosis and relieve biliary obstruction, endoscopic biopsy of the duodenal papilla was first performed, followed by endoscopic dual biliary and pancreatic duct stent placement. Histopathological examination revealed severe glandular dysplasia in the duodenal papilla with focal features suspicious for malignant transformation. Positron emission ACCEPTED tomography-computed tomography (PET-CT) demonstrated increased fluorodeoxyglucose (FDG) uptake in the distal common bile duct (suggesting malignancy) and the portal vein (indicating tumor thrombus). After multidisciplinary team (MDT) evaluation, a diagnosis of ampullary carcinoma with PVTT was established. The patient declined neoadjuvant chemotherapy. She underwent laparoscopic pancreatoduodenectomy (LPD) with portal vein resection and reconstruction (PVRR). Intraoperatively, a 2-cm segment of the portal vein containing tumor thrombus was resected, and end-to-end anastomosis was performed (Figure 2A-B). Frozen section analysis confirmed malignancy within the portal vein thrombus (Figure 3). Final pathological findings revealed a moderately differentiated adenocarcinoma (2.0×2.0×1.8 cm) in the ampullary region, with no evidence of metastatic involvement in the regional lymph nodes. Tumor thrombus was identified in the portal vein. The patient recovered uneventfully and was discharged on postoperative day 18. She subsequently completed four cycles of nab-paclitaxel plus gemcitabine (nab-P/G) chemotherapy and remains disease-free at 6 months of follow-up. 3. Discussion PVTT is a common complication of hepatocellular carcinoma (HCC) and significantly impacts prognosis and clinical staging. While PVTT is well-documented in HCC, pancreatic [3 4] cancer, gastric cancer, and colorectal cancer , its occurrence in ampullary carcinoma has not been previously reported. To our knowledge, this represents the first case of PVTT secondary to ampullary carcinoma. ACCEPTED Ampullary carcinomas arise from the ampulla of Vater, a confluence of the bile duct, pancreatic duct, and duodenal mucosa. These tumors account for 0.2% of gastrointestinal malignancies and typically originate from adenomatous or dysplastic precursors. The 5-year [5] overall survival ranges from 35% to 50% . Per the National Comprehensive Cancer Network (NCCN) guidelines, ampullary carcinomas are classified into pancreatobiliary, intestinal, or [6] mixed subtypes, with intestinal subtypes demonstrating superior prognosis .In this case, the patient demonstrated full-thickness duodenal wall invasion with pancreatic extension and biliary duct involvement, consistent with pancreatobiliary-type ampullary carcinoma.This pathophysiological correlation elucidates the preoperative histopathological findings of focal suspicious malignant characteristics in the duodenal papilla. [7] PVTT classification systems include the Japanese Vp classification and Cheng’s [8] classification . Using Cheng’s criteria, this case represents Type III PVTT (main portal vein involvement). Given the absence of prior reports on ampullary carcinoma with PVTT, our MDT advocated for aggressive surgical management (LPD + PVRR) followed by adjuvant nab-P/G chemotherapy, achieving favorable short-term outcomes. Regarding the formation of PVTT in this case, we speculate that it may be related to CTCs. CTCs are cancer cells in circulation dissociated from primary tumors. CTCs can be used as a [9] biomarker to noninvasively monitor cancer progression and guide therapy . CTCs are considered to be an active source of metastases due to their potential stem cell features and epithelial‐ mesenchymal transition (EMT) traits, which allow them to disseminate effectively. From the perspective of the tumor microenvironment, the generation of CTC is a part of the transformation of extracellular matrix (ECM) and EMT. CTCs obtain mesenchymal features via EMT, infiltrate the ECM by releasing proteolytic degradation enzymes, and cross the basement ACCEPTED membrane. Eventually, they enter circulation and extravasate to form secondary micro- [10] metastasis . However, it cannot be confirmed whether the PVTT in this case originated from CTCs, and how CTCs survive and form PVTT in the portal vein remains unknown. In summary, we report a rare case of a patient with ampullary carcinoma who developed a tumor thrombus in the portal vein without liver metastasis. We speculate that this translocation may be related to CTCs, but whether PVTT originates from CTCs and how CTCs survive without matrix support remains unknown. ACCEPTED References [1] Voutsadakis IA, Doumas S, Tsapakidis K, Papagianni M, Papandreou CN. Bone and brain metastases from ampullary adenocarcinoma. World J Gastroenterol. 2009 Jun 7;15(21):2665-8. [2] Tsai YE, Chien KH, Li YF, Lai SW. Rare Orbital Metastasis Originating from Ampullary Adenocarcinoma. Medicina (Kaunas). 2021 Nov 12;57(11):1238. [3] Liang Ong SC, Batumaly SK, Jusoh SM. Portal vein tumor thrombus from gastric cancer. J Ultrason. 2018;18(75):365-368. [4] Shintani D, Tajima Y, Baba H, Matsuzawa T, Kumamoto K, Kumagai Y, Ozawa F, Ishibashi K, Haga N, Ishida H. [Colon cancer with portal vein tumor thrombosis-a case report and review of the literature]. Gan To Kagaku Ryoho. 2012 Nov;39(12):2243-5. Japanese. [5] Chiorean EG, Chiaro MD, Tempero MA, Malafa MP, Benson AB, Cardin DB, Christensen JA, Chung V, Czito B, Dillhoff M, Donahue TR, Dotan E, Fountzilas C, Glazer ES, Hardacre J, Hawkins WG, Klute K, Ko AH, Kunstman JW, LoConte N, Lowy AM, Masood A, Moravek C, Nakakura EK, Narang AK, Nardo L, Obando J, Polanco PM, Reddy S, Reyngold M, Scaife C, Shen J, Truty MJ, Vollmer C, Wolff RA, Wolpin BM, Rn BM, Lubin S, Darlow SD. Ampullary Adenocarcinoma, Version 1.2023, NCCN Clinical Practice Guidelines in Oncology. J Natl Compr Canc Netw. 2023 Jul;21(7):753-782. [6] Chang DK, Jamieson NB, Johns AL, Scarlett CJ, Pajic M, Chou A, Pinese M, Humphris JL, Jones MD, Toon C, Nagrial AM, Chantrill LA, Chin VT, Pinho AV, Rooman I, Cowley MJ, Wu J, Mead RS, Colvin EK, Samra JS, Corbo V, Bassi C, Falconi M, Lawlor RT, Crippa S, Sperandio N, Bersani S, Dickson EJ, Mohamed MA, Oien KA, Foulis AK, Musgrove EA, Sutherland RL, Kench JG, Carter CR, Gill AJ, Scarpa A, McKay CJ, Biankin AV. Histomolecular phenotypes and outcome in adenocarcinoma of the ampulla of vater. J Clin Oncol. 2013 Apr 1;31(10):1348-56. ACCEPTED [7] Ikai I, Yamamoto Y, Yamamoto N, Terajima H, Hatano E, Shimahara Y, Yamaoka Y. Results of hepatic resection for hepatocellular carcinoma invading major portal and/or hepatic veins. Surg Oncol Clin N Am. 2003 Jan;12(1):65-75, ix. [8] Shi J, Lai EC, Li N, Guo WX, Xue J, Lau WY, Wu MC, Cheng SQ. A new classification for hepatocellular carcinoma with portal vein tumor thrombus. J Hepatobiliary Pancreat Sci. 2011 Jan;18(1):74-80. [9] Lin D, Shen L, Luo M, Zhang K, Li J, Yang Q, Zhu F, Zhou D, Zheng S, Chen Y, Zhou J. Circulating tumor cells: biology and clinical significance. Signal Transduct Target Ther. 2021 Nov 22;6(1):404. [10] Li Z, Zhao M, Qi X, Tang Y, Cheng S. Mechanisms of portal vein tumour thrombus formation and development in patients with hepatocellular carcinoma. J Cell Mol Med. 2023 Aug;27(15):2103-2111. ACCEPTED Figure 1: Pancreatic enhanced CT: (A) An ill-defined density is observed in the distal common bile duct with no apparent enhancement ; (B) Portal vein shows filling defect with low density. Figure 2: Introperative imaging: (A) The portal vein tumor thrombus in the main portal vein of the portal vein. (B) The portal vein after reconstruction is completed. Figure 3: Macroscopic specimen of the portal vein ACCEPTED Figure 1 ACCEPTED Figure 2f ACCEPTED Figure 3 ACCEPTED http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Pancreatology Wolters Kluwer Health

Portal Vein Tumor Thrombus Originating from Ampullary Adenocarcinoma: A Case Report

Cao, Ye; Li, Qiyuan; Zhang, Binru; Wang, Yongwei; Sun, Bei; Chen, Hua
Journal of Pancreatology , Volume Publish Ahead of Print – Jul 3, 2025
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Wolters Kluwer Health
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Copyright © 2025 The Chinese Medical Association, Published by Wolters Kluwer Health, Inc.
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2577-3577
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2096-5664
DOI
10.1097/jp9.0000000000000226
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Abstract

Journal of Pancreatology DOI: 10.1097/JP9.0000000000000226 Portal Vein Tumor Thrombus Originating from Ampullary Adenocarcinoma: A Case Report Ye Cao, Qiyuan Li, Binru Zhang, Yongwei Wang, Bei Sun, Hua Chen Department of Pancreatic and Biliary Surgery, The First Affiliated Hospital of Harbin Medical University, Harbin, China Corresponding Author: Hua Chen (Email:[email protected]) Authors’ contributions Ye Cao, Qiyuan Li, Binru Zhang wrote the manuscript. Yongwei Wang, Bei Sun, Hua Chen performed the surgery. All authors conceived the study, participated in its design and coordination, and helped to draft the manuscript. All authors have read and approved the final manuscript. Consent for publication Written informed consent was obtained from the patient for publication of this case report and any accompanying images. Ethical Approval This case report represents a fully anonymized retrospective presentation. Therefore, the need for ethical approval was waived according to the regulation of the Ethics Committee of The First Affiliated Hospital of Harbin Medical University. This case report was not registered in any online public registration platform. ACCEPTED Competing interests The authors declare that they have no competing interests. This is an open access article distributed under the terms of the Creative Commons Attribution- Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. ACCEPTED Abstract: We present a rare case of a 62-year-old female with ampullary adenocarcinoma complicated by portal vein tumor thrombus (PVTT). The patient presented with upper abdominal pain, jaundice, and fever. Imaging revealed a distal common bile duct mass with portal vein thrombosis. After multidisciplinary discussion, she underwent laparoscopic pancreaticoduodenectomy with portal vein resection and reconstruction. Histopathology confirmed the presence of adenocarcinoma and PVTT. The patient had an uneventful recovery and is disease-free at 6 months post-surgery. Keywords: Ampullary carcinoma; Portal vein tumor thrombus; Metastases ACCEPTED 1. Introduction Ampullary carcinoma is a rare malignant tumor of the gastrointestinal tract, classified as a type of peri-ampullary cancer. Common metastatic sites include the liver, peritoneum, and lungs. [1] Additionally, there are reports of ampullary carcinoma metastasizing to the bones, brain , and [2] orbit . Portal vein tumor thrombosis (PVTT) in ampullary carcinoma is exceedingly rare, and a systematic literature search conducted across PubMed, EMBASE, and Web of Science databases confirmed that no cases of this specific tumor entity have been reported in the English-language medical literature to date. We present the first documented case of PVTT associated with ampullary carcinoma. 2. Case Report A 62-year-old female presented with a one-week history of upper abdominal pain accompanied by fever, jaundice, nausea, and abdominal distension. Six months prior, she had undergone laparoscopic common bile duct exploration for gallbladder and bile duct stones. Admission laboratory tests revealed: ALT: 115 U/L, AST: 122 U/L, TBIL: 107.6 μmol/L, DBIL: 87.9 μmol/L, and CA19-9: 99.71 U/mL. Magnetic resonance cholangiopancreatography (MRCP) demonstrated a distal common bile duct stone with secondary intrahepatic and extrahepatic bile duct dilation. Contrast-enhanced pancreatic CT revealed a mass in the distal common bile duct causing low bile duct obstruction and thrombosis in the main portal vein (Figure 1A-B). To confirm the diagnosis and relieve biliary obstruction, endoscopic biopsy of the duodenal papilla was first performed, followed by endoscopic dual biliary and pancreatic duct stent placement. Histopathological examination revealed severe glandular dysplasia in the duodenal papilla with focal features suspicious for malignant transformation. Positron emission ACCEPTED tomography-computed tomography (PET-CT) demonstrated increased fluorodeoxyglucose (FDG) uptake in the distal common bile duct (suggesting malignancy) and the portal vein (indicating tumor thrombus). After multidisciplinary team (MDT) evaluation, a diagnosis of ampullary carcinoma with PVTT was established. The patient declined neoadjuvant chemotherapy. She underwent laparoscopic pancreatoduodenectomy (LPD) with portal vein resection and reconstruction (PVRR). Intraoperatively, a 2-cm segment of the portal vein containing tumor thrombus was resected, and end-to-end anastomosis was performed (Figure 2A-B). Frozen section analysis confirmed malignancy within the portal vein thrombus (Figure 3). Final pathological findings revealed a moderately differentiated adenocarcinoma (2.0×2.0×1.8 cm) in the ampullary region, with no evidence of metastatic involvement in the regional lymph nodes. Tumor thrombus was identified in the portal vein. The patient recovered uneventfully and was discharged on postoperative day 18. She subsequently completed four cycles of nab-paclitaxel plus gemcitabine (nab-P/G) chemotherapy and remains disease-free at 6 months of follow-up. 3. Discussion PVTT is a common complication of hepatocellular carcinoma (HCC) and significantly impacts prognosis and clinical staging. While PVTT is well-documented in HCC, pancreatic [3 4] cancer, gastric cancer, and colorectal cancer , its occurrence in ampullary carcinoma has not been previously reported. To our knowledge, this represents the first case of PVTT secondary to ampullary carcinoma. ACCEPTED Ampullary carcinomas arise from the ampulla of Vater, a confluence of the bile duct, pancreatic duct, and duodenal mucosa. These tumors account for 0.2% of gastrointestinal malignancies and typically originate from adenomatous or dysplastic precursors. The 5-year [5] overall survival ranges from 35% to 50% . Per the National Comprehensive Cancer Network (NCCN) guidelines, ampullary carcinomas are classified into pancreatobiliary, intestinal, or [6] mixed subtypes, with intestinal subtypes demonstrating superior prognosis .In this case, the patient demonstrated full-thickness duodenal wall invasion with pancreatic extension and biliary duct involvement, consistent with pancreatobiliary-type ampullary carcinoma.This pathophysiological correlation elucidates the preoperative histopathological findings of focal suspicious malignant characteristics in the duodenal papilla. [7] PVTT classification systems include the Japanese Vp classification and Cheng’s [8] classification . Using Cheng’s criteria, this case represents Type III PVTT (main portal vein involvement). Given the absence of prior reports on ampullary carcinoma with PVTT, our MDT advocated for aggressive surgical management (LPD + PVRR) followed by adjuvant nab-P/G chemotherapy, achieving favorable short-term outcomes. Regarding the formation of PVTT in this case, we speculate that it may be related to CTCs. CTCs are cancer cells in circulation dissociated from primary tumors. CTCs can be used as a [9] biomarker to noninvasively monitor cancer progression and guide therapy . CTCs are considered to be an active source of metastases due to their potential stem cell features and epithelial‐ mesenchymal transition (EMT) traits, which allow them to disseminate effectively. From the perspective of the tumor microenvironment, the generation of CTC is a part of the transformation of extracellular matrix (ECM) and EMT. CTCs obtain mesenchymal features via EMT, infiltrate the ECM by releasing proteolytic degradation enzymes, and cross the basement ACCEPTED membrane. Eventually, they enter circulation and extravasate to form secondary micro- [10] metastasis . However, it cannot be confirmed whether the PVTT in this case originated from CTCs, and how CTCs survive and form PVTT in the portal vein remains unknown. In summary, we report a rare case of a patient with ampullary carcinoma who developed a tumor thrombus in the portal vein without liver metastasis. We speculate that this translocation may be related to CTCs, but whether PVTT originates from CTCs and how CTCs survive without matrix support remains unknown. ACCEPTED References [1] Voutsadakis IA, Doumas S, Tsapakidis K, Papagianni M, Papandreou CN. Bone and brain metastases from ampullary adenocarcinoma. World J Gastroenterol. 2009 Jun 7;15(21):2665-8. [2] Tsai YE, Chien KH, Li YF, Lai SW. Rare Orbital Metastasis Originating from Ampullary Adenocarcinoma. Medicina (Kaunas). 2021 Nov 12;57(11):1238. [3] Liang Ong SC, Batumaly SK, Jusoh SM. Portal vein tumor thrombus from gastric cancer. J Ultrason. 2018;18(75):365-368. [4] Shintani D, Tajima Y, Baba H, Matsuzawa T, Kumamoto K, Kumagai Y, Ozawa F, Ishibashi K, Haga N, Ishida H. [Colon cancer with portal vein tumor thrombosis-a case report and review of the literature]. Gan To Kagaku Ryoho. 2012 Nov;39(12):2243-5. Japanese. [5] Chiorean EG, Chiaro MD, Tempero MA, Malafa MP, Benson AB, Cardin DB, Christensen JA, Chung V, Czito B, Dillhoff M, Donahue TR, Dotan E, Fountzilas C, Glazer ES, Hardacre J, Hawkins WG, Klute K, Ko AH, Kunstman JW, LoConte N, Lowy AM, Masood A, Moravek C, Nakakura EK, Narang AK, Nardo L, Obando J, Polanco PM, Reddy S, Reyngold M, Scaife C, Shen J, Truty MJ, Vollmer C, Wolff RA, Wolpin BM, Rn BM, Lubin S, Darlow SD. Ampullary Adenocarcinoma, Version 1.2023, NCCN Clinical Practice Guidelines in Oncology. J Natl Compr Canc Netw. 2023 Jul;21(7):753-782. [6] Chang DK, Jamieson NB, Johns AL, Scarlett CJ, Pajic M, Chou A, Pinese M, Humphris JL, Jones MD, Toon C, Nagrial AM, Chantrill LA, Chin VT, Pinho AV, Rooman I, Cowley MJ, Wu J, Mead RS, Colvin EK, Samra JS, Corbo V, Bassi C, Falconi M, Lawlor RT, Crippa S, Sperandio N, Bersani S, Dickson EJ, Mohamed MA, Oien KA, Foulis AK, Musgrove EA, Sutherland RL, Kench JG, Carter CR, Gill AJ, Scarpa A, McKay CJ, Biankin AV. Histomolecular phenotypes and outcome in adenocarcinoma of the ampulla of vater. J Clin Oncol. 2013 Apr 1;31(10):1348-56. ACCEPTED [7] Ikai I, Yamamoto Y, Yamamoto N, Terajima H, Hatano E, Shimahara Y, Yamaoka Y. Results of hepatic resection for hepatocellular carcinoma invading major portal and/or hepatic veins. Surg Oncol Clin N Am. 2003 Jan;12(1):65-75, ix. [8] Shi J, Lai EC, Li N, Guo WX, Xue J, Lau WY, Wu MC, Cheng SQ. A new classification for hepatocellular carcinoma with portal vein tumor thrombus. J Hepatobiliary Pancreat Sci. 2011 Jan;18(1):74-80. [9] Lin D, Shen L, Luo M, Zhang K, Li J, Yang Q, Zhu F, Zhou D, Zheng S, Chen Y, Zhou J. Circulating tumor cells: biology and clinical significance. Signal Transduct Target Ther. 2021 Nov 22;6(1):404. [10] Li Z, Zhao M, Qi X, Tang Y, Cheng S. Mechanisms of portal vein tumour thrombus formation and development in patients with hepatocellular carcinoma. J Cell Mol Med. 2023 Aug;27(15):2103-2111. ACCEPTED Figure 1: Pancreatic enhanced CT: (A) An ill-defined density is observed in the distal common bile duct with no apparent enhancement ; (B) Portal vein shows filling defect with low density. Figure 2: Introperative imaging: (A) The portal vein tumor thrombus in the main portal vein of the portal vein. (B) The portal vein after reconstruction is completed. Figure 3: Macroscopic specimen of the portal vein ACCEPTED Figure 1 ACCEPTED Figure 2f ACCEPTED Figure 3 ACCEPTED

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Journal of Pancreatology – Wolters Kluwer Health

Published: Jul 3, 2025

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