Case Report: Epstein-Barr virus negative lymphoepithelioma-like cholangiocarcinoma: a rare tumor that deserves further exploration. Report of a case with distinct genomic and clinical features

Lymphoepithelioma-like cholangiocarcinoma (LEL-CC) is a rare variant of cholangiocarcinoma characterized by undifferentiated malignant epithelial cells and a significant lymphoid infiltrate. Due to its rarity, there is currently no established treatment protocol for LEL-CC, and limited data are avai...

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Main Authors: Salvatore Corallo, Chiara Milani, Alessandro Vanoli, Anna Gallotti, Anna Pagani, Francesco Serra, Marcello Maestri, Valentina Ravetta, Angioletta Lasagna, Paolo Pedrazzoli, Francesco Agustoni
Format: Article
Language:English
Published: Frontiers Media S.A. 2025-08-01
Series:Frontiers in Oncology
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Online Access:https://www.frontiersin.org/articles/10.3389/fonc.2025.1620328/full
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author Salvatore Corallo
Salvatore Corallo
Chiara Milani
Alessandro Vanoli
Alessandro Vanoli
Anna Gallotti
Anna Pagani
Francesco Serra
Marcello Maestri
Valentina Ravetta
Angioletta Lasagna
Paolo Pedrazzoli
Paolo Pedrazzoli
Francesco Agustoni
Francesco Agustoni
author_facet Salvatore Corallo
Salvatore Corallo
Chiara Milani
Alessandro Vanoli
Alessandro Vanoli
Anna Gallotti
Anna Pagani
Francesco Serra
Marcello Maestri
Valentina Ravetta
Angioletta Lasagna
Paolo Pedrazzoli
Paolo Pedrazzoli
Francesco Agustoni
Francesco Agustoni
author_sort Salvatore Corallo
collection DOAJ
description Lymphoepithelioma-like cholangiocarcinoma (LEL-CC) is a rare variant of cholangiocarcinoma characterized by undifferentiated malignant epithelial cells and a significant lymphoid infiltrate. Due to its rarity, there is currently no established treatment protocol for LEL-CC, and limited data are available regarding the genomic landscape of this rare tumor. In this report, we present the case of an 84-year-old woman with a history of Hepatitis C-related cirrhosis who was diagnosed with EBV-negative LEL-CC. This tumor presented as a double primary tumor together with a moderately differentiated hepatocellular carcinoma, at the time of first diagnosis. After surgical resection, the patient experienced a relapse with intracolonic metastasis. Comprehensive genomic profiling revealed unique genetic features consistent with LEL carcinoma of other sites of origin. The genetic and clinical characteristics of our case highlight the need for further research on this rare variant of cholangiocarcinoma. Gaining insights into the molecular mechanisms behind this type of cancer could lead to the development of effective targeted therapies or immunotherapeutic approaches.
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institution Kabale University
issn 2234-943X
language English
publishDate 2025-08-01
publisher Frontiers Media S.A.
record_format Article
series Frontiers in Oncology
spelling doaj-art-b0c2a8ac92a5438dbacaba58cf3e07e12025-08-20T03:43:02ZengFrontiers Media S.A.Frontiers in Oncology2234-943X2025-08-011510.3389/fonc.2025.16203281620328Case Report: Epstein-Barr virus negative lymphoepithelioma-like cholangiocarcinoma: a rare tumor that deserves further exploration. Report of a case with distinct genomic and clinical featuresSalvatore Corallo0Salvatore Corallo1Chiara Milani2Alessandro Vanoli3Alessandro Vanoli4Anna Gallotti5Anna Pagani6Francesco Serra7Marcello Maestri8Valentina Ravetta9Angioletta Lasagna10Paolo Pedrazzoli11Paolo Pedrazzoli12Francesco Agustoni13Francesco Agustoni14Department of Internal Medicine and Medical Therapy, University of Pavia, Pavia, ItalyUnit of Oncology, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyDepartment of Internal Medicine and Medical Therapy, University of Pavia, Pavia, ItalyDepartment of Molecular Medicine, University of Pavia, Pavia, ItalyUnit of Anatomic Pathology, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyInstitute of Radiology, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyUnit of Oncology, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyUnit of Oncology, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyDivision of General Surgery 1, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyGastroenterology and Endoscopy Unit, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyUnit of Oncology, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyDepartment of Internal Medicine and Medical Therapy, University of Pavia, Pavia, ItalyUnit of Oncology, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyDepartment of Internal Medicine and Medical Therapy, University of Pavia, Pavia, ItalyUnit of Oncology, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyLymphoepithelioma-like cholangiocarcinoma (LEL-CC) is a rare variant of cholangiocarcinoma characterized by undifferentiated malignant epithelial cells and a significant lymphoid infiltrate. Due to its rarity, there is currently no established treatment protocol for LEL-CC, and limited data are available regarding the genomic landscape of this rare tumor. In this report, we present the case of an 84-year-old woman with a history of Hepatitis C-related cirrhosis who was diagnosed with EBV-negative LEL-CC. This tumor presented as a double primary tumor together with a moderately differentiated hepatocellular carcinoma, at the time of first diagnosis. After surgical resection, the patient experienced a relapse with intracolonic metastasis. Comprehensive genomic profiling revealed unique genetic features consistent with LEL carcinoma of other sites of origin. The genetic and clinical characteristics of our case highlight the need for further research on this rare variant of cholangiocarcinoma. Gaining insights into the molecular mechanisms behind this type of cancer could lead to the development of effective targeted therapies or immunotherapeutic approaches.https://www.frontiersin.org/articles/10.3389/fonc.2025.1620328/fulllymphoepithelioma-like cholangiocarcinomaHCC-CC double primary tumorintracolonic metastasisEBV-negative cholangiocarcinomaHCV-related cirrhosis
spellingShingle Salvatore Corallo
Salvatore Corallo
Chiara Milani
Alessandro Vanoli
Alessandro Vanoli
Anna Gallotti
Anna Pagani
Francesco Serra
Marcello Maestri
Valentina Ravetta
Angioletta Lasagna
Paolo Pedrazzoli
Paolo Pedrazzoli
Francesco Agustoni
Francesco Agustoni
Case Report: Epstein-Barr virus negative lymphoepithelioma-like cholangiocarcinoma: a rare tumor that deserves further exploration. Report of a case with distinct genomic and clinical features
Frontiers in Oncology
lymphoepithelioma-like cholangiocarcinoma
HCC-CC double primary tumor
intracolonic metastasis
EBV-negative cholangiocarcinoma
HCV-related cirrhosis
title Case Report: Epstein-Barr virus negative lymphoepithelioma-like cholangiocarcinoma: a rare tumor that deserves further exploration. Report of a case with distinct genomic and clinical features
title_full Case Report: Epstein-Barr virus negative lymphoepithelioma-like cholangiocarcinoma: a rare tumor that deserves further exploration. Report of a case with distinct genomic and clinical features
title_fullStr Case Report: Epstein-Barr virus negative lymphoepithelioma-like cholangiocarcinoma: a rare tumor that deserves further exploration. Report of a case with distinct genomic and clinical features
title_full_unstemmed Case Report: Epstein-Barr virus negative lymphoepithelioma-like cholangiocarcinoma: a rare tumor that deserves further exploration. Report of a case with distinct genomic and clinical features
title_short Case Report: Epstein-Barr virus negative lymphoepithelioma-like cholangiocarcinoma: a rare tumor that deserves further exploration. Report of a case with distinct genomic and clinical features
title_sort case report epstein barr virus negative lymphoepithelioma like cholangiocarcinoma a rare tumor that deserves further exploration report of a case with distinct genomic and clinical features
topic lymphoepithelioma-like cholangiocarcinoma
HCC-CC double primary tumor
intracolonic metastasis
EBV-negative cholangiocarcinoma
HCV-related cirrhosis
url https://www.frontiersin.org/articles/10.3389/fonc.2025.1620328/full
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