A bile duct lesion originating from intrahepatic bile ducts is generally regarded as an incidental pathologic finding in liver specimens. However, a recent study on the molecular classification of intrahepatic cholangiocarcinoma has focused on the heterogeneity of this carcinoma and has suggested that the cells of different origins present in the biliary tree may have a major role in the mechanism of oncogenesis. In this review, benign intrahepatic bile duct lesions—regarded in the past as reactive changes or remnant developmental anomalies and now noted to have potential for developing precursor lesions of intrahepatic cholangiocarcinoma—are discussed by focusing on the histopathologic features and its implications in clinical practice.
Citations
Citations to this article as recorded by
Spectrum of Biliary Lesions/Neoplasms in Hepatic Parenchyma with Reference to a Precursor of Small Duct-Type Intrahepatic Cholangiocarcinoma: Comprehensive Categorization into Three Groups Yasuni Nakanuma, Motoko Sasaki, Yuko Kakuda, Takuma Oishi Cancers.2026; 18(2): 328. CrossRef
Comprehensive molecular-clinical profiling of cholangiocarcinoma according to pathologic subtypes Keun Soo Ahn, Daniel O'Brien, Yoo Na Kang, Taofic Mounajjed, Hye Won Lee, Yong Hoon Kim, Tae-Seok Kim, Min Jae Kim, Jin-Yi Han, Mitesh J. Borad, Lewis R. Roberts HPB.2026; 28(6): 796. CrossRef
A deceptively benign biliary lesion revealing an ALK‐rearranged intrahepatic cholangiocarcinoma Anne‐Cécile Brunac, Marie Danjoux, Hadrien Reboul, Ronan Guillemin, Fabrice Muscari, Janick Selves Histopathology.2026; 89(1): 189. CrossRef
Intrahepatic Bile Duct Adenoma with a History of Asbestos Exposure: a Case Report Mitsuru Yamakawa, Tomoo Kobayashi, Fumie Nakayama, Noriyuki Iwama Kanzo.2026; 67(2): 126. CrossRef
Spectrum of Biliary and Nonbiliary Neoplasms Growing and Spreading Within the Lumen of the Bile Ducts Yasuni Nakanuma, Yasunori Sato, Yuko Kakuda, Takuma Oishi Cancers.2026; 18(9): 1356. CrossRef
Biliary Adenofibroma and the Threat of Malignant Transformation Mohamed A. Youssef, Joseph Gosnell, Heather L. Stevenson International Journal of Surgical Pathology.2025; 33(7): 1658. CrossRef
Neoplastic ductal plate malformation pattern rather than von Meyenburg complexes may be an early neoplastic lesion of small duct-type intrahepatic cholangiocarcinoma Yasuni Nakanuma, Motoko Sasaki, Yuko Kakuda, Kenichi Harada, Yasunori Sato, Takashi Sugino Human Pathology.2025; 161: 105865. CrossRef
Biliary pseudo tumor associated with hepatic atrophy Albina Joldoshova, Binny Khandakar, Hwajeong Lee, Robert Lam, Dhanpat Jain Human Pathology.2025; 163: 105879. CrossRef
Histopathologische Diagnostik von soliden und zystischen Pankreasläsionen mit Hauptaugenmerk auf dem duktalen Adenokarzinom Irene Esposito, Aslihan Yavas, Lena Häberle Die Pathologie.2024; 45(1): 5. CrossRef
Recent Advances in Pathology of Intrahepatic Cholangiocarcinoma Joon Hyuk Choi, Swan N. Thung Cancers.2024; 16(8): 1537. CrossRef
INTRAHEPATIC BILIARY PROLIFERATIONS: HISTOPATHOLOGY AND POTENTIAL IMMUNOHISTOCHEMICAL MARKERS André Bubna HIRAYAMA, Evandro Sobroza de MELLO, Venâncio Avancini Ferreira ALVES Arquivos de Gastroenterologia.2023; 60(3): 393. CrossRef
Development and Validation of a Difficulty Scoring System for Laparoscopic Liver Resection to Treat Hepatolithiasis Yeongsoo Jo, Jai Young Cho, Ho-Seong Han, Yoo-Seok Yoon, Hae Won Lee, Jun Suh Lee, Boram Lee, Eunhye Lee, Yeshong Park, MeeYoung Kang, Junghyun Lee Medicina.2022; 58(12): 1847. CrossRef
Validation of a difficulty scoring system for laparoscopic liver resection in hepatolithiasis Jinju Kim, Jai Young Cho, Ho-Seong Han, Yoo-Seok Yoon, YoungRok Choi, Jun Suh Lee, Boram Lee, Junyub Kim Surgical Endoscopy.2021; 35(3): 1148. CrossRef
Composite MRI Task Construction from CT Images based on Deep Convolution Neural Network Liang Min, Yi Gu, Rui Xue, Yi Ren, Bo Gao Journal of Imaging Science and Technology.2021; 65(3): 030404-1. CrossRef
Re-Recognizing the Cellular Origin of the Primary Epithelial Tumors of the Liver Jiliang Feng, Ruidong Zhu, Yu Yin, Shanshan Wang, Lei Zhou, Fudong Lv, Dawei Zhao Journal of Hepatocellular Carcinoma.2021; Volume 8: 1537. CrossRef
Clinicopathological characteristics of intrahepatic cholangiocarcinoma according to gross morphologic type: cholangiolocellular differentiation traits and inflammation- and proliferation-phenotypes Taek Chung, Hyungjin Rhee, Ji Hae Nahm, Youngsic Jeon, Jeong Eun Yoo, Young-Joo Kim, Dai Hoon Han, Young Nyun Park HPB.2020; 22(6): 864. CrossRef
Biliary Intraepithelial Neoplasia in Non-biliary Cirrhosis—Report From 100 Explanted Livers: A Single Center Experience Maryam Zarei, Alireza Shasaeefar, Kurosh Kazemi, Masoud Dehghani, Seyed-Ali Malekhosseini, Bita Geramizadeh Clinical Pathology.2019;[Epub] CrossRef
Intrahepatic cholangiocarcinoma after Fontan procedure in an adult with visceral heterotaxy Dehua Wang, Darren Marshall, Gruschen Veldtman, Anita Gupta, Andrew T. Trout, Juan Villafane, Kevin Bove Pathology - Research and Practice.2018; 214(6): 914. CrossRef
Human Chorionic Plate-Derived Mesenchymal Stem Cells Restore Hepatic Lipid Metabolism in a Rat Model of Bile Duct Ligation Yun Bin Lee, Jong Ho Choi, Eun Nam Kim, Jin Seok, Hyun-Jung Lee, Jung-Hwan Yoon, Gi Jin Kim Stem Cells International.2017; 2017: 1. CrossRef
Surgery is the preferred treatment for bile duct hamartomas Xiao-Yu Yang, Hai-Bo Zhang, Bin Wu, Ai-Jun Li, Xiao-Hui Fu Molecular and Clinical Oncology.2017; 7(4): 649. CrossRef
Undifferentiated embryonal sarcoma of the liver (UESL) is rare primary hepatic sarcoma and is known to occur in pediatric patients. This case is the UESL occurred in a 51-year old male patient. Multilocular cystic lesion was composed of primitive spindle cells without specific differentiation. This rare case would help to review differential diagnosis of primary sarcoma in liver and cystic neoplasm of the liver.
Citations
Citations to this article as recorded by
Case Report: Diagnosis and treatment of an undifferentiated embryonal sarcoma of the liver Xiaojiao Qiu, Lexing Zhang, Fan Sun Frontiers in Surgery.2025;[Epub] CrossRef
Unusual presentation of an embryonal sarcoma of the liver in an adult Adarsh Barwad, Sameer Rastogi, S H Chandrashekhara, Prasenjit Das Indian Journal of Cancer.2025; 62(3): 439. CrossRef
Case Report: Adult UESL mimicking a simple hepatic cyst and initially misdiagnosed as a benign lesion Yuanhang Mo, Jinrong Feng, Chunhui Liang, Xiaoqing Ma, Zongquan Wu, Xingwang Su, Shan He, Xiaoyou Xu, Yongfeng Liao, Yongdong Liu Frontiers in Oncology.2025;[Epub] CrossRef
Undifferentiated embryonal sarcoma of the liver masquerading as a cystadenoma in a young adult: a case report Yanfang Huang, Xiao He, Linyu Shangguan, Yuli Zhou, Lexing Zhang, Yipai Wu Journal of Medical Case Reports.2024;[Epub] CrossRef
Undifferentiated Embryonal Sarcoma of the Liver: First Case Reported in Central America Amilcar Antonio Alfaro García, Ludwing Bacon Fonseca, Maria del Rosario Romero Alvarez Cancer Reports.2024;[Epub] CrossRef
Primäre Lebersarkome – Empfehlungen zur chirurgischen Therapie Eva Maria Dobrindt, Robert Öllinger, Anne Flörcken, Sven Märdian, Moritz Schmelzle, Georg Lurje, Johann Pratschke, Wenzel Schoening Zentralblatt für Chirurgie - Zeitschrift für Allgemeine, Viszeral-, Thorax- und Gefäßchirurgie.2023; 148(02): 165. CrossRef
Undifferentiated embryonal sarcoma of the liver: a case report of a rare case in an adult patient Ana M Marques, Guilherme Fontinha, Maria João Ferreira, Milene Sá, Júlio Constantino, Jorge Pereira Journal of Surgical Case Reports.2023;[Epub] CrossRef
Undifferentiated embryonal sarcoma of the liver presenting as a hemorrhagic mass in a pregnant woman Yang Jiang, Frank Chen, Jordan LeGout, Lauren Alexander, John Stauffer, Allie Metcalfe Clinical Imaging.2021; 77: 58. CrossRef
Undifferentiated Embryonal Sarcoma of the Liver in Children Versus Adults: A National Cancer Database Analysis Ioannis A. Ziogas, Irving J. Zamora, Harold N. Lovvorn III, Christina E. Bailey, Sophoclis P. Alexopoulos Cancers.2021; 13(12): 2918. CrossRef
Long‐term survival outcomes of undifferentiated embryonal sarcoma of the liver: a pooled analysis of 308 patients Zhiyong Wu, Youzhu Wei, Zhibin Cai, Yanming Zhou ANZ Journal of Surgery.2020; 90(9): 1615. CrossRef
Undifferentiated Embryonal Sarcoma of the Liver Identified after the Initial Diagnosis of a Hepatic Cyst Yusaku Manabe, Haruki Uojima, Hisashi Hidaka, Xue Shao, Shuichiro Iwasaki, Naohisa Wada, Kousuke Kubota, Yoshiaki Tanaka, Takahide Nakazawa, Akitaka Shibuya, Masaaki Ichinoe, Yusuke Kumamoto, Takashi Kaizu, Wasaburo Koizumi Internal Medicine.2020; 59(19): 2375. CrossRef
Undifferentiated embryonal sarcoma of the liver: Clinical characteristics and outcomes Chong Zhang, Chang-Jun Jia, Can Xu, Qiu-Ju Sheng, Xiao-Guang Dou, Yang Ding World Journal of Clinical Cases.2020; 8(20): 4763. CrossRef
Undifferentiated embryonal sarcoma of liver in an adult with spontaneous rupture and tumour thrombus in the right atrium Narendra Pandit, Lokesh S. Jaiswal, Vijay Shrestha, Lalijan Awale, Shailesh Adhikary ANZ Journal of Surgery.2019;[Epub] CrossRef
Hanging Undifferentiated Embryonal Sarcoma of the Liver in Adult: an Unusual Presentation of an Aggressive Tumor Narendra Pandit, Kunal Bikram Deo, Lokesh Shekher Jaiswal, Anju Pradhan, Shailesh Adhikary Journal of Gastrointestinal Cancer.2019; 50(3): 689. CrossRef
Undifferentiated Embryonal Sarcoma of the Liver: a Great Masquerader Alexander M. Sy, Maureen Whitsett, Xiaodong Li, Neil D. Theise, Nabil N. Dagher, Sonja Olsen Journal of Gastrointestinal Cancer.2019; 50(4): 1043. CrossRef
Child with hepatic hydatid cyst: A diagnostic uncertainty Shashi Ranjani, Intezar Mehdi, Basant Mahadevappa, Shivakumar Swamy Indian Journal of Child Health.2019; 6(1): 46. CrossRef
Undifferentiated embryonal sarcoma of the liver in adult patient: A report of two cases Germán Esteban Sánchez-Morales, Uriel Emilio Clemente-Gutiérrez, Francisco Emmanuel Alvarez-Bautista, Oscar Santes-Jasso, Paulina Carpinteyro-Espin, Miguel Angel Mercado Annals of Hepato-Biliary-Pancreatic Surgery.2018; 22(3): 269. CrossRef
A case of adult undifferentiated embryonal sarcoma of the liver successfully treated with right trisectionectomy: a case report Akiko Mori, Koji Fukase, Kunihiro Masuda, Naoaki Sakata, Masamichi Mizuma, Hideo Ohtsuka, Takanori Morikawa, Kei Nakagawa, Hiroki Hayashi, Fuyuhiko Motoi, Takeshi Naitoh, Keigo Murakami, Michiaki Unno Surgical Case Reports.2017;[Epub] CrossRef
A Rare Case of Hepatocellular Carcinoma With Osteoclast-Like Giant Cells: Favorable Outcome Following Early Surgical Resection Ryohei Aoyama, Kentaro Yasuchika, Masaki Yamada, Naoto Nakamura, Satoshi Yokoyama Cureus.2025;[Epub] CrossRef
Undifferentiated carcinoma of the liver with osteoclast-like giant cells: a case report and literature review Lixia Lu, Li Wang, Can Peng, Li Chen, Ximan He, Chenning Shao, Chunnian Wang, Rong Ge Infectious Agents and Cancer.2024;[Epub] CrossRef
Osteoclast-like giant cells in hepatocellular carcinoma case description and review of the literature Anke H. C. Gielen, Iryna Samarska, Marcel Den Dulk, Jan Beckervordersandforth, Kees H. C. Dejong, Stefan A. W. Bouwense, Maxime Dewulf Acta Chirurgica Belgica.2023; 123(2): 178. CrossRef
Undifferentiated hepatic carcinoma with osteoclast-like giant cells: A case report and literature review Yujiao Deng, Ya Wang, Yan Zhang, Na Yang, Xingli Ji, Bing Wu Frontiers in Oncology.2023;[Epub] CrossRef
Imaging features of primary hepatic sarcomatoid carcinoma: Differentiation from hepatocellular carcinoma and intrahepatic cholangiocarcinoma on CT: A preliminary study Lintao Chen, Shijian Ruan, Pan Wang, Yongna Cheng, Yubizhuo Wang, Wuwei Tian, Hongbin Zhang, Xiuming Zhang, Wenjie Liang Heliyon.2023; 9(3): e14123. CrossRef
Advances in Histological and Molecular Classification of Hepatocellular Carcinoma Joon Hyuk Choi, Swan N. Thung Biomedicines.2023; 11(9): 2582. CrossRef
Sarcomatoid hepatocellular carcinoma versus conventional hepatocellular carcinoma: a systematic review and meta-analysis Tian-Run Lv, Hai-Jie Hu, Parbatraj Regmi, Fei Liu, Fu-Yu Li Journal of Cancer Research and Clinical Oncology.2022; 148(7): 1685. CrossRef
Recurrent hepatocellular carcinoma with osteoclast-like giant cells: a case report Mone Tsukimoto, Kazushi Sugimoto, Ryuta Shigefuku, Ryosuke Sugimoto, Hiroto Yuasa, Katsunori Uchida, Norihiko Yamamoto Journal of Medical Case Reports.2022;[Epub] CrossRef
Clinical Manifestations and Outcomes of Patients with Sarcomatoid Hepatocellular Carcinoma Sih‐Han Liao, Tung‐Hung Su, Yung‐Ming Jeng, Po‐Chin Liang, Ding‐Shinn Chen, Chien‐Hung Chen, Jia‐Horng Kao Hepatology.2019; 69(1): 209. CrossRef
Management and outcomes among patients with sarcomatoid hepatocellular carcinoma: A population‐based analysis Lu Wu, Diamantis I. Tsilimigras, Ayesha Farooq, J. Madison Hyer, Katiuscha Merath, Anghela Z. Paredes, Rittal Mehta, Kota Sahara, Feng Shen, Timothy M. Pawlik Cancer.2019; 125(21): 3767. CrossRef
The prognosis of subsequent surgical treatment in patients with sarcomatoid carcinoma in the liver: A retrospective study Anna Kan, Rong-ping Guo International Journal of Surgery.2018; 55: 145. CrossRef
A Case of Hepatic Angiosarcoma with Osteoclast-like Giant Cells Takahiro MIZUI, Keigo MURAKAMI, Yasuyuki HARA, Shigehito MIYAGI, Takeshi NAITOH, Michiaki UNNO, Takashi KAMEI Nihon Rinsho Geka Gakkai Zasshi (Journal of Japan Surgical Association).2018; 79(2): 382. CrossRef
Intrahepatic sarcomatoid cholangiocarcinoma Sara Sintra, Rui Costa, Carlos Filipe, Adélia Simão BMJ Case Reports.2018; 2018: bcr-2018-225017. CrossRef
Morphologic Subtypes of Hepatocellular Carcinoma Michael S. Torbenson Gastroenterology Clinics of North America.2017; 46(2): 365. CrossRef
Sarcomatoid cholangiocarcinoma with osteoclast-like giant cells associated with hepatolithiasis: A case report Hye Min Kim, Haeryoung Kim, Young Nyun Park Clinical and Molecular Hepatology.2015; 21(3): 309. CrossRef
First
Prev
