S3222 An Unusual Presentation of Unresectable Hilar Cholangiocarcinoma in a Young Adult: A Case of Hemosuccus Pancreaticus

Introduction: Cholangiocarcinoma is a rare malignancy with peak incidence in the seventh decade of life. We report a highly atypical case of unresectable hilar cholangiocarcinoma in a previously healthy 32-year-old man who presented with an unusual and life-threatening manifestation. This case highlights an exceptionally rare presentation of hilar cholangiocarcinoma manifesting as hemosuccus pancreaticus—a phenomenon scarcely reported in literature. The patient’s age and the bleeding presentation defy classical teaching and underscore the diagnostic challenges associated with cholangiocarcinoma in young adults. Case Description/Methods: Patient initially presented to outpatient clinic for evaluation of abdominal pain and progressive jaundice with cholestatic liver enzyme elevation. No prior hepatobiliary or gastrointestinal disease. In the midst of workup, patient presented to emergency room for syncope, melena and symptomatic anemia. Patient was tachycardic and hypotensive on admission with a hemoglobin downtrend from 14.2 g/dl to 7.4 g/dl. Patient was admitted to intensive care unit for GI hemorrhage. Esophagogastroduodenoscopy revealed active oozing of blood from the ampulla of Vater, raising suspicion for hemosuccus pancreaticus, a rare cause of upper GI bleeding due to hemorrhage through the pancreatic duct. Patient underwent magnetic resonance cholangiopancreatography (MRCP) which demonstrated an infiltrative hilar mass with intrahepatic biliary ductal dilation. Endoscopic retrograde cholangiopancreatography (ERCP) with biliary stenting of the hilar stricture was performed to relieve obstruction. Endoscopic ultrasound (EUS) revealed a hypoechoic perihilar mass, and fine needle aspiration yielded atypical cells suspicious for malignancy. Following multidisciplinary discussion and comprehensive imaging review, the lesion was deemed unresectable hilar cholangiocarcinoma and referral to a liver transplant center for further evaluation. Discussion: Clinicians should maintain a high index of suspicion for biliary tract malignancies even in younger patients with unexplained cholestatic liver patterns and hyperbilirubinemia, especially when standard workup reveals atypical findings such as ampullary bleeding. This case adds to the growing but limited body of literature on early-onset cholangiocarcinoma and introduces a unique, life-threatening presentation. Recognition of atypical symptoms is critical for timely diagnosis, multidisciplinary management, and consideration for liver transplant in unresectable cases.

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Publication Details

Journal
The American Journal of Gastroenterology
Published
2025-10-01
DOI
https://doi.org/10.14309/01.ajg.0001140348.85398.53
Primary Topic
Cholangiocarcinoma and Gallbladder Cancer Studies
Type
article
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0.00
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article

S3222 An Unusual Presentation of Unresectable Hilar Cholangiocarcinoma in a Young Adult: A Case of Hemosuccus Pancreaticus

Andrew Shehata, Devjit S. Nayar, Adwait Patel
The American Journal of Gastroenterology
Cholangiocarcinoma and Gallbladder Cancer Studies
article

S3222 An Unusual Presentation of Unresectable Hilar Cholangiocarcinoma in a Young Adult: A Case of Hemosuccus Pancreaticus

Andrew Shehata, Devjit S. Nayar, Adwait Patel
article en

Abstract

Introduction: Cholangiocarcinoma is a rare malignancy with peak incidence in the seventh decade of life. We report a highly atypical case of unresectable hilar cholangiocarcinoma in a previously healthy 32-year-old man who presented with an unusual and life-threatening manifestation. This case highlights an exceptionally rare presentation of hilar cholangiocarcinoma manifesting as hemosuccus pancreaticus—a phenomenon scarcely reported in literature. The patient’s age and the bleeding presentation defy classical teaching and underscore the diagnostic challenges associated with cholangiocarcinoma in young adults. Case Description/Methods: Patient initially presented to outpatient clinic for evaluation of abdominal pain and progressive jaundice with cholestatic liver enzyme elevation. No prior hepatobiliary or gastrointestinal disease. In the midst of workup, patient presented to emergency room for syncope, melena and symptomatic anemia. Patient was tachycardic and hypotensive on admission with a hemoglobin downtrend from 14.2 g/dl to 7.4 g/dl. Patient was admitted to intensive care unit for GI hemorrhage. Esophagogastroduodenoscopy revealed active oozing of blood from the ampulla of Vater, raising suspicion for hemosuccus pancreaticus, a rare cause of upper GI bleeding due to hemorrhage through the pancreatic duct. Patient underwent magnetic resonance cholangiopancreatography (MRCP) which demonstrated an infiltrative hilar mass with intrahepatic biliary ductal dilation. Endoscopic retrograde cholangiopancreatography (ERCP) with biliary stenting of the hilar stricture was performed to relieve obstruction. Endoscopic ultrasound (EUS) revealed a hypoechoic perihilar mass, and fine needle aspiration yielded atypical cells suspicious for malignancy. Following multidisciplinary discussion and comprehensive imaging review, the lesion was deemed unresectable hilar cholangiocarcinoma and referral to a liver transplant center for further evaluation. Discussion: Clinicians should maintain a high index of suspicion for biliary tract malignancies even in younger patients with unexplained cholestatic liver patterns and hyperbilirubinemia, especially when standard workup reveals atypical findings such as ampullary bleeding. This case adds to the growing but limited body of literature on early-onset cholangiocarcinoma and introduces a unique, life-threatening presentation. Recognition of atypical symptoms is critical for timely diagnosis, multidisciplinary management, and consideration for liver transplant in unresectable cases.

The American Journal of GastroenterologyVol. 120(10S2)
Openalex Percentile: Top 41%
Cholangiocarcinoma and Gallbladder Cancer Studies
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