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Shin SH, Koh YS, Song S. Hepatic epithelioid hemangioendothelioma managed with minimally invasive surgery: A case report. World J Clin Cases 2025; 13:104924. [DOI: 10.12998/wjcc.v13.i22.104924] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/06/2025] [Revised: 03/22/2025] [Accepted: 04/14/2025] [Indexed: 05/14/2025] Open
Abstract
BACKGROUND Hepatic epithelioid hemangioendothelioma (HEHE) is a rare malignant vascular liver tumor diagnosed by histopathological evaluation. Standardized treatment is challenging because of its rarity; hepatectomy is preferred for solitary lesions and multiple transplantations. There is no consensus on the optimal treatment for HEHE; however, surgical excision is often considered effective. This report presents a case of initially suspected cholangiocarcinoma or renal cell carcinoma (RCC) metastasis, which was later confirmed as HEHE, with no recurrence during follow-up.
CASE SUMMARY A 52-year-old man with a history of left nephrectomy for RCC presented with an incidentally detected liver mass and nonspecific abdominal discomfort. Imaging revealed a 3-cm centripetal enhancing lesion in the right hepatic dome with indeterminate malignant potential. The patient underwent a laparoscopic right anterior sectionectomy and remained recurrence-free without complications during the 3-year follow-up period.
CONCLUSION Managing HEHE is challenging. Accurate diagnosis and surgical options, such as resection or transplantation, are essential with tailored multidisciplinary follow-up. The authors have read the CARE Checklist (2016) and the manuscript was prepared and revised according to the CARE Checklist (2016).
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Affiliation(s)
- Sang Hoon Shin
- Department of General Surgery, Chonnam National University Hwasun Hospital, Hwasun 519-763, South Korea
| | - Yang Seok Koh
- Department of General Surgery, Chonnam National University Hwasun Hospital, Chonnam National University Medical School, Hwasun 519-763, South Korea
| | - Sanghwa Song
- Department of General Surgery, Chonnam National University Hospital, Gwangju 5681, South Korea
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Stanietzky N, Salem AE, Elsayes KM, Rezvani M, Palmquist S, Ahmed I, Marey A, Faria S, Gaballah AH, Menias CO, Shaaban AM. Unusual liver tumors: spectrum of imaging findings with pathologic correlation. Diagn Interv Radiol 2025; 31:58-67. [PMID: 38856323 PMCID: PMC11880871 DOI: 10.4274/dir.2024.242827] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/25/2024] [Accepted: 05/18/2024] [Indexed: 06/11/2024]
Abstract
The liver is a common location for both primary and secondary cancers of the abdomen. Radiologists become familiar with the typical imaging features of common benign and malignant liver tumors; however, many types of liver tumors are encountered infrequently. Due to the rarity of these lesions, their typical imaging patterns may not be easily recognized, meaning their underlying pathologic features may not be discovered or suggested until an invasive biopsy is performed. In this review article, we discuss multiple hepatic neoplasms that are both unusual and rare. Some have typical imaging patterns, whereas others are non-specific and can only be included in the differential diagnosis. The clinical history and serologic findings are often critical in suggesting these entities; therefore, these are also discussed to familiarize the radiologist with the appropriate clinical setting of each. The article includes an image-rich description of each entity with accompanying figures describing the ultrasonography, computed tomography, and magnetic resonance imaging features of each disease process. Novel therapies and prognosis of several of the diseases are also included in the discussion.
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Affiliation(s)
- Nir Stanietzky
- University of Texas MD Anderson Cancer Center Department of Radiology, Texas, USA
| | - Ahmed Ebada Salem
- The University of Utah Department of Radiology, Utah, USA
- Alexandria University Faculty of Medicine Department of Radiodiagnosis and Intervention, Alexandria, Egypt
| | - Khaled M. Elsayes
- University of Texas MD Anderson Cancer Center Department of Radiology, Texas, USA
| | - Maryam Rezvani
- The University of Utah Department of Radiology, Utah, USA
| | - Sarah Palmquist
- University of Texas MD Anderson Cancer Center Department of Radiology, Texas, USA
| | - Imran Ahmed
- University of Texas MD Anderson Cancer Center Department of Radiology, Texas, USA
| | - Ahmed Marey
- Alexandria University Faculty of Medicine Department of Radiodiagnosis and Intervention, Alexandria, Egypt
| | - Silvana Faria
- University of Texas MD Anderson Cancer Center Department of Radiology, Texas, USA
| | - Ayman H. Gaballah
- University of Texas MD Anderson Cancer Center Department of Radiology, Texas, USA
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3
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Malik M, Idrees RB, Mirza Z, Anwar S, Ahmad B, Chaudhary MH. Rare Encounter With Hepatic Epithelioid Hemangioendothelioma: A Case Report. Cureus 2025; 17:e80567. [PMID: 40225499 PMCID: PMC11994122 DOI: 10.7759/cureus.80567] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 03/14/2025] [Indexed: 04/15/2025] Open
Abstract
Malignant hepatic epithelioid hemangioendothelioma (HEHE) is a rare vascular tumor with variable malignant potential, often affecting middle-aged individuals. Hepatic involvement is uncommon, presenting diagnostic challenges due to overlapping imaging features with other liver pathologies. We report a case of a 35-year-old female with HEHE initially presenting with epigastric pain and an incidental liver mass. Imaging revealed multifocal hepatic lesions, confirmed as HEHE through biopsy and immunohistochemistry. Despite initial stability on tamoxifen, disease progression occurred after seven years, manifesting as an increase in the size and number of hepatic lesions. Hepatic transplantation was planned, which was refused by the patient. Subsequently, the disease progressed to a large confluent hepatic mass with calcifications, lymphadenopathy, and pulmonary metastases. Systemic chemotherapy was henceforth initiated. This case underscores the importance of a multimodal approach integrating imaging, histopathology, and tailored therapeutic strategies for diagnosing and managing HEHE. Early detection and comprehensive management are critical to improving outcomes where liver transplantation offers a potential cure.
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Affiliation(s)
- Mariam Malik
- Department of Radiology, Nuclear Medicine, Oncology and Radiotherapy Institute-Atomic Energy Cancer Hospital (NORI-AECH), Islamabad, PAK
| | - Rana Bilal Idrees
- Department of Radiology, Institute of Nuclear Medicine and Oncology-Atomic Energy Cancer Hospital (INMOL-AECH), Lahore, PAK
| | - Zeeshan Mirza
- Department of Radiology, Institute of Nuclear Medicine and Oncology-Atomic Energy Cancer Hospital (INMOL-AECH), Lahore, PAK
| | - Sadia Anwar
- Department of Radiology, Institute of Nuclear Medicine and Oncology-Atomic Energy Cancer Hospital (INMOL-AECH), Lahore, PAK
| | - Barira Ahmad
- Department of Radiology, Institute of Nuclear Medicine and Oncology-Atomic Energy Cancer Hospital (INMOL-AECH), Lahore, PAK
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Svintsitskiy V, Tsip N, Nespryadko S, Samokhvalova O, Khoptiana O, Yeriushkin D, Krotevych M, Trokhymych S, Skoroda L. Ovarian Epithelioid Hemangioendothelioma. Case Report. Exp Oncol 2025; 46:402-407. [PMID: 39985344 DOI: 10.15407/exp-oncology.2024.04.402] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/20/2025] [Indexed: 02/24/2025]
Abstract
Epithelioid hemangioendothelioma is a rare vascular sarcoma commonly occurring in the liver, soft tissues, and organs of the chest cavity. This study provides a detailed description of the clinical presentation, diagnosis, and treatment of epithelioid hemangioendothelioma of the ovary in a 71-year-old patient. According to the pathohistological examination, a mesenchymal tumor of the ovary with epithelioid cells was diagnosed. The immunohistochemical tests confirmed the diagnosis of epithelioid hemangioendothelioma. This clinical case is of interest for clinical practitioners due to the rare location of the tumor in female reproductive organs, which has not been previously documented in the medical literature.
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Affiliation(s)
- V Svintsitskiy
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
| | - N Tsip
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
| | - S Nespryadko
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
| | - O Samokhvalova
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
| | - O Khoptiana
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
| | - D Yeriushkin
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
| | - M Krotevych
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
| | - S Trokhymych
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
| | - L Skoroda
- State Non-Commercial Enterprise «National Cancer Institute», Kyiv, Ukraine
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Wu D, Chen Z, Wu Z, Lin J. Rare hepatic epithelioid hemangioendothelioma with popliteal fossa metastases: A case report. Asian J Surg 2024:S1015-9584(24)02880-X. [PMID: 39665926 DOI: 10.1016/j.asjsur.2024.11.231] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/09/2024] [Accepted: 11/25/2024] [Indexed: 12/13/2024] Open
Affiliation(s)
- Dingwei Wu
- Department of Orthopedics, Fujian Medical University Union Hospital, Fuzhou, Fujian, 350001, China.
| | - Zhi Chen
- Department of Orthopedics, Fujian Medical University Union Hospital, Fuzhou, Fujian, 350001, China.
| | - Zhengru Wu
- Department of Orthopedics, Fujian Medical University Union Hospital, Fuzhou, Fujian, 350001, China.
| | - Jiajun Lin
- Department of Orthopedics, Fujian Medical University Union Hospital, Fuzhou, Fujian, 350001, China.
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Atyah MM, Sun Y, Yang Z. The challenges of hepatic epithelioid hemangioendothelioma: the diagnosis and current treatments of a problematic tumor. Orphanet J Rare Dis 2024; 19:449. [PMID: 39616351 PMCID: PMC11608485 DOI: 10.1186/s13023-024-03354-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/02/2023] [Accepted: 09/04/2024] [Indexed: 12/06/2024] Open
Abstract
BACKGROUND Hepatic epithelioid hemangioendothelioma (HEHE) is a malignant vascular tumor known for its rarity. The different types of this hepatic tumor (single, multiple-nodular or diffused) indicate different prognosis and treatment plans. However, the heterogenic clinical manifestation creates a dilemma and a wide range of challenges when attending to HEHE patients. This review addresses the unique profile and clinical challenges that complicate the diagnosis and treatment of HEHE while focusing on current therapeutic strategies and their limitations. MAIN TEXT The unclear etiology is a challenging feature of HEHE. The exact involvement of potential risk factors and mechanism of development are still undefined. Relevant genetic alterations like WWTR1-CAMTA1 fusion have been investigated; however, they are only applicable as diagnostic markers and their influence on therapeutic efficacy is largely unknown. Other characteristics include asymptomatic manifestation, lack of unique hepatic functional alterations, high rates of misdiagnosis and late-stage identification when metastases already exist. Currently, tissue biopsy is the main tool to establish a definite diagnosis but is challenged with the limited awareness to suspect HEHE at early stages and the lack of relevant guidelines due to the rarity and the insufficiency of relevant research. The absence of treatment guidelines is the greatest challenge of HEHE. Generally, Surgical approaches are recommended due to the benefits of prolonged survival and enhanced prognosis. Nonetheless, only a minority of patients are eligible for resections while liver transplants are faced with severe insufficiency of donor organs and long wait-lists. On the other hand, a variety of non-surgical treatments (like anti-angiogenic agents, interferon alpha-2B and sirolimus) are presented with a promising potential. However, relevant studies are challenged with limited sample-sizes and lack of prospective designs. CONCLUSION Regardless to decades passing since its discovery, HEHE still creates a dilemma due to its challenging clinical profile and lack of treatment guidelines. Raising awareness of HEHE in clinical practices improves the ability to diagnose this rare tumor at early stages and develop stronger research strategies and treatment guidelines to regulate the medical care provided to HEHE patients.
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Affiliation(s)
- Manar Mikhail Atyah
- Department of Hepatobiliary and Pancreatic Surgery, China-Japan Friendship Hospital, Beijing, China
| | - Yongliang Sun
- Department of Hepatobiliary and Pancreatic Surgery, China-Japan Friendship Hospital, Beijing, China.
| | - Zhiying Yang
- Department of Hepatobiliary and Pancreatic Surgery, China-Japan Friendship Hospital, Beijing, China.
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Pan A, Xu S, Hong L, Zhu W, Zhou Y, Wang X. Hepatic epithelioid Hemangioendothelioma presenting synchronously with hepatocellular carcinoma-a case report. Oxf Med Case Reports 2024; 2024:omae140. [PMID: 39588500 PMCID: PMC11586779 DOI: 10.1093/omcr/omae140] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/10/2024] [Revised: 08/23/2024] [Accepted: 09/06/2024] [Indexed: 11/27/2024] Open
Abstract
BACKGROUNDS Primary hepatic epithelioid hemangioendothelioma (HEHE) is a rare neoplasm of vascular origin with varying biologic behavior, making it challenging to diagnose. CASE PRESENTATION We present a case of synchronous hepatocellular carcinoma (HCC) and HEHE in a 43-year-old Chinese male patient. Multiple hypoechoic liver lesions were depicted, but no specific imaging findings were detected on enhanced computed tomography (CT) or contrast-enhanced magnetic resonance imaging (MRI). The patient then underwent [18F]-FDG PET/CT, [11C]-acetate PET/CT, and [68Ga]Ga-FAPI-04 PET/CT. The HEHE lesions demonstrated no uptake on both 18F-FDG and 11C-acetate PET/CT imaging, but presented a clear visualization in [68Ga]Ga-FAPI-04 PET/CT. The largest lesion located in segment VII was finally diagnosed as HCC, while the other smaller ones were diagnosed as HEHE, which was confirmed by immunohistochemical staining for CD31. To the best of our knowledge, only 2 cases have been reported in the worldwide literature, and the first case undertook both 11C-acetate and [68Ga]Ga-FAPI-04 PET/CT instead of 18F-FDG PET/CT. CONCLUSION In this report, we show that HCC and HEHE may occur synchronously, and HEHE should be considered when liver lesions are detected. [68Ga]Ga-FAPI-04 PET/CT has great potential in the detection, staging and therapy selection of HEHE.
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Affiliation(s)
- Anna Pan
- Department of Infectious Diseases, The Third Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang, China
| | - Shuang Xu
- Department of Infectious Diseases, The Third Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang, China
| | - Liang Hong
- Department of Infectious Diseases, The Third Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang, China
| | - Wenwei Zhu
- Department of General Surgery, Huashan Hospital Affiliated to Fudan University, Shanghai, China
| | - Yu Zhou
- Department of Infectious Diseases, The Third Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang, China
| | - Xiaoyang Wang
- Department of Radiology, The Third Affiliated Hospital of Wenzhou Medical University, Wenzhou, Zhejiang, China
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Liu X, Yang P, Liu L, Si S, Zhou R, Liu T, Tan H. Long-term prognosis and treatment modalities of hepatic epithelioid hemangioendothelioma: a retrospective study of 228 patients. BMC Cancer 2024; 24:1285. [PMID: 39415114 PMCID: PMC11481724 DOI: 10.1186/s12885-024-13053-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/16/2024] [Accepted: 10/10/2024] [Indexed: 10/18/2024] Open
Abstract
BACKGROUND Hepatic epithelioid hemangioendothelioma (EHE) is an extremely rare tumour. The aim of this study was to investigate the long-term prognosis and its relationship with treatment modalities. METHODS From March 2014 to June 2024, a total of 234 patients with histologically confirmed hepatic EHE were treated or followed up regularly by our team. The patients' clinical data at the time of diagnosis and initial treatment modalities were retrospectively collected. Kaplan-Meier curves were constructed to determine overall survival (OS). To explore prognostic factors and treatment outcomes, univariable and multivariable Cox proportional hazard models were developed. RESULTS A total of 228 patients were ultimately included. The median age of the cohort was 41 years. For all patients, the OS of 1-, 3- and 5-year were 96.2%, 87.9% and 84.9%, respectively. For patients who underwent liver transplantation (LT), the OS of 1- and 3-year were 62.5% and 25%, respectively. No difference was found in the OS between patients who received surgical therapy and those who did not (1-year: 100% vs. 96.9%; 3-year: 90.1% vs. 91.5%; 5-year: 87.2% vs. 88.2%; P = 0.891). In the multivariable analysis, age ≥ 60 years [HR (95% CI): 4.207 (1.266-13.973), P = 0.019], the size of the largest lesion > 10 cm [HR (95% CI): 12.140 (1.419-103.872), P = 0.023] and LT [HR (95% CI): 5.502 (1.343-22.536), P = 0.018] were poor prognostic factors. CONCLUSIONS Compared with nonsurgical therapy, surgical therapy has no advantage in terms of long-term survival. The role of LT in the management of hepatic EHE should be reevaluated. Age ≥ 60 years and the size of the largest lesion > 10 cm are poor prognostic factors.
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Affiliation(s)
- Xiaolei Liu
- Second Department of Hepatopancreatobiliary Surgery, China-Japan Friendship Hospital, 2 Yinghua Dongjie, Hepingli, Beijing, 100029, China.
| | - Peijun Yang
- Second Department of Hepatopancreatobiliary Surgery, China-Japan Friendship Hospital, 2 Yinghua Dongjie, Hepingli, Beijing, 100029, China
| | - Liguo Liu
- Second Department of Hepatopancreatobiliary Surgery, China-Japan Friendship Hospital, 2 Yinghua Dongjie, Hepingli, Beijing, 100029, China
| | - Shuang Si
- Second Department of Hepatopancreatobiliary Surgery, China-Japan Friendship Hospital, 2 Yinghua Dongjie, Hepingli, Beijing, 100029, China
| | - Ruiquan Zhou
- Second Department of Hepatopancreatobiliary Surgery, China-Japan Friendship Hospital, 2 Yinghua Dongjie, Hepingli, Beijing, 100029, China
| | - Tiantong Liu
- Second Department of Hepatopancreatobiliary Surgery, China-Japan Friendship Hospital, 2 Yinghua Dongjie, Hepingli, Beijing, 100029, China
| | - Haidong Tan
- Second Department of Hepatopancreatobiliary Surgery, China-Japan Friendship Hospital, 2 Yinghua Dongjie, Hepingli, Beijing, 100029, China.
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Mao Y, Liu W, Yao Y, Zou C, Tian B. A young male with primary hepatic epithelioid hemangioendothelioma. REVISTA ESPANOLA DE ENFERMEDADES DIGESTIVAS 2024. [PMID: 39267490 DOI: 10.17235/reed.2024.10733/2024] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 09/17/2024]
Abstract
Epithelioid hemangioendothelioma (EHEs) is a vascular endothelial-derived tumor, which can be seen in the liver, mediastinum, lung and other parts, and most often occurs in the liver. Hepatic epithelioid hemangioendothelioma (HEHE) is rare in clinical practice. The etiology and pathogenesis of HEHE are still unclear, and it depends on imaging and pathological examination. A case of multiple space-occupying lesions in the liver was reported. The patient had no history of chronic liver disease, and was diagnosed as hepatic epithelioid hemangioendothelioma by abdominal ultrasonography，contrast-enhanced CT，and PET-CT. Partial hepatectomy and intraoperative microwave ablation were performed. Postoperative pathological diagnosis confirmed the diagnosis of hepatic epithelioid hemangioendothelioma. This case report aims to provide clinicians with ideas for the diagnosis and treatment of the disease.
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Affiliation(s)
- Yuxuan Mao
- Hepatobiliary Surgery, The First Affiliated Hospital of Hunan Normal University, China
| | - Weinan Liu
- Hepatobiliary Surgery, The First Affiliated Hospital of Hunan Normal University, China
| | - Yuping Yao
- Hepatobiliary Surgery, The First Affiliated Hospital of Hunan Normal University, China
| | - Chen Zou
- Hepatobiliary Surgery, The First Affiliated Hospital of Hunan Normal University, China
| | - Binzhang Tian
- Hepatobiliary Surgery, The First Affiliated Hospital of Hunan Normal University, China
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10
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Jakipov M, Karimov A, Khamitova Z, Kochiev B, Bolatov D, Spatayev Z, Bolsynbekova S, Kuanysh Z, Zharlyganova D. MRI and PET/CT of multiple primary hepatic angiosarcomas in 25-year-old man: A case report. J Clin Imaging Sci 2024; 14:32. [PMID: 39246734 PMCID: PMC11380813 DOI: 10.25259/jcis_69_2024] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/17/2024] [Accepted: 07/19/2024] [Indexed: 09/10/2024] Open
Abstract
This study aims to provide a comprehensive understanding of primary hepatic angiosarcoma, a rare and aggressive malignancy, focusing on its diagnostic challenges and unique imaging characteristics. The objective is to delineate the distinctive features of angiosarcoma through computed tomography and magnetic resonance imaging modalities, contributing to improved diagnostic precision and adding valuable insights to the scientific literature. We present the case of a 25-year-old male with primary hepatic angiosarcoma, emphasizing the challenges in distinguishing it from other vascular tumors.
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Affiliation(s)
- Murat Jakipov
- Department of Radiology, National Research Oncology Center, Astana, Kazakhstan
| | - Aslan Karimov
- Department of Radiology, National Research Oncology Center, Astana, Kazakhstan
| | - Zaukiya Khamitova
- Department of Science, National Research Oncology Center, Astana, Kazakhstan
| | - Bayram Kochiev
- Department of Radiology, National Research Oncology Center, Astana, Kazakhstan
| | - Dauren Bolatov
- Department of Radiology, National Research Oncology Center, Astana, Kazakhstan
| | - Zhanat Spatayev
- Department of Hepatopancreatobiliary Surgery, Onco-hepatology and Organ Transplantation, National Research Oncology Center, Astana, Kazakhstan
| | | | - Zhuldyz Kuanysh
- Department of Science, National Research Oncology Center, Astana, Kazakhstan
| | - Dinara Zharlyganova
- Department of Science, National Research Oncology Center, Astana, Kazakhstan
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Frenette C, Mendiratta-Lala M, Salgia R, Wong RJ, Sauer BG, Pillai A. ACG Clinical Guideline: Focal Liver Lesions. Am J Gastroenterol 2024; 119:1235-1271. [PMID: 38958301 DOI: 10.14309/ajg.0000000000002857] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/07/2023] [Accepted: 04/25/2024] [Indexed: 07/04/2024]
Abstract
Focal liver lesions (FLLs) have become an increasingly common finding on abdominal imaging, especially asymptomatic and incidental liver lesions. Gastroenterologists and hepatologists often see these patients in consultation and make recommendations for management of multiple types of liver lesions, including hepatocellular adenoma, focal nodular hyperplasia, hemangioma, and hepatic cystic lesions including polycystic liver disease. Malignancy is important to consider in the differential diagnosis of FLLs, and healthcare providers must be familiar with the diagnosis and management of FLLs. This American College of Gastroenterology practice guideline uses the best evidence available to make diagnosis and management recommendations for the most common FLLs.
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Affiliation(s)
| | | | - Reena Salgia
- Department of Gastroenterology/Hepatology, Henry Ford Health, Detroit, Michigan, USA
| | - Robert J Wong
- Division of Gastroenterology and Hepatology, Veterans Affairs Palo Alto Health Care System and Stanford University School of Medicine, Palo Alto, California, USA
| | - Bryan G Sauer
- Division of Gastroenterology and Hepatology, University of Virginia, Charlottesville, Virginia, USA
| | - Anjana Pillai
- Division of Gastroenterology, Hepatology, and Nutrition, Department of Medicine, University of Chicago Medical Center, University of Chicago, Chicago, Illinois, USA
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12
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Jevdokimova N, Jevdokimov D, Ivanova J, Isajevs S, Staka A, Pukitis A. A Young Female with a Hepatic Epithelioid Hemangioendothelioma. Eur J Case Rep Intern Med 2024; 11:004529. [PMID: 38984174 PMCID: PMC11229466 DOI: 10.12890/2024_004529] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/01/2024] [Accepted: 04/02/2024] [Indexed: 07/11/2024] Open
Abstract
Background Primary hepatic epithelioid hemangioendothelioma (HEHE) is an extremely rare tumour of vascular origin with an incidence of <0.1 cases per 100,000 people worldwide. Case description A 29-year-old female with the history of epigastric pain and unintentional weight loss (3 kg over six months) was referred for upper endoscopy. The examination was without visual pathological findings, but a rapid urease test was positive. First-line treatment with clarithromycin-containing triple therapy for Helicobacter pylori infection was given. After completion of eradication therapy, diffuse abdominal pain developed. An abdominal computed tomography (CT) showed multiple liver nodules. Three consecutive core liver biopsies were performed and were inconclusive. A subsequent surgical liver nodule resection was performed. Histopathology of the specimen revealed grade 2 hepatocellular carcinoma; bone scintigraphy was negative for metastasis. A multidisciplinary team (MDT) recommended giving the patient sorafenib, which was poorly tolerated. The histology was reviewed using immunohistochemistry staining at the request of the oncologist, which showed expression of CD31 and CD34. Based on clinical, morphological and immunohistochemistry findings, a diagnosis of hepatic epithelioid hemangioendothelioma was made. Based on the multidisciplinary team's findings, liver transplantation was indicated as the only curative treatment. Conclusion Because of the rarity of this disease, combining clinical, radiological and histopathological methods as well as an MDT approach can help to reach the correct final diagnosis. As demonstrated in this clinical case, it is crucial to perform immunohistochemistry of a liver biopsy to confirm a HEHE diagnosis. LEARNING POINTS Hepatic epithelioid hemangioendothelioma is a rare vascular tumour that is often misdiagnosed and mismanaged.This case emphasises the critical importance of interdisciplinary teamwork and the use of non-invasive and invasive techniques to achieve a definitive diagnosis.
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Affiliation(s)
- Natalija Jevdokimova
- Department of Internal Medicine, Pauls Stradins Clinical University Hospital, Riga, Latvia
- University of
Latvia, Faculty of Medicine, Riga, Latvia
| | - Denis Jevdokimov
- University of
Latvia, Faculty of Medicine, Riga, Latvia
- Centre of Gastroenterology, Hepatology and Nutrition, Pauls Stradins Clinical University Hospital, Riga, Latvia
| | - Jelena Ivanova
- Centre of Gastroenterology, Hepatology and Nutrition, Pauls Stradins Clinical University Hospital, Riga, Latvia
| | | | - Aiga Staka
- University of
Latvia, Faculty of Medicine, Riga, Latvia
- Centre of Gastroenterology, Hepatology and Nutrition, Pauls Stradins Clinical University Hospital, Riga, Latvia
| | - Aldis Pukitis
- University of
Latvia, Faculty of Medicine, Riga, Latvia
- Centre of Gastroenterology, Hepatology and Nutrition, Pauls Stradins Clinical University Hospital, Riga, Latvia
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13
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Muscatello LV, Massari F, Roccabianca P, Sarli G, Benazzi C, Bianchi ML. Hepatic Epithelioid Hemangioendothelioma in a Dog. Animals (Basel) 2024; 14:1302. [PMID: 38731306 PMCID: PMC11083527 DOI: 10.3390/ani14091302] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/25/2024] [Revised: 04/22/2024] [Accepted: 04/23/2024] [Indexed: 05/13/2024] Open
Abstract
A 5-year-old spayed female Breton dog was referred for a thyroid nodule. A total body CT scan evidenced multifocal hepatic nodules. Cytological liver samples were hemodiluted and non-diagnostic. Following a thyroidectomy, the histology was consistent with a follicular-compact thyroid carcinoma. On laparoscopy, most hepatic lobes had multifocal dark-red nodules that were biopsied for histology. Microscopically, the hepatic parenchyma in the nodules was substituted by blood channels lined by bland spindle cells but adjacent to epithelioid neoplastic cells, single or in clusters, embedded in a moderate amount of edematous collagen matrix. These cells had optically empty cytoplasmic space, occasionally containing erythrocytes (microlumina). Spindle and epithelioid cells expressed membranous-to-cytoplasmic CD31 and FVIII-RA consistent with endothelial origin. Based on morphology and immunolabelling, a hemangioendothelioma with epithelioid differentiation was diagnosed. Lesions in the liver were initially stable, showing progression with time. The dog was alive with no systemic clinical signs 36 months after laparoscopy.
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Affiliation(s)
- Luisa Vera Muscatello
- Department of Veterinary Medical Sciences, University of Bologna, Via Tolara di Sopra, 50, Ozzano dell’Emilia, 40064 Bologna, Italy; (G.S.); (C.B.)
| | - Federico Massari
- DOCVET Clinica Veterinaria Nervianese, Via Rho 2, 20014 Nerviano, Italy; (F.M.); (M.L.B.)
| | - Paola Roccabianca
- Department of Veterinary Medicine, University of Milano, via dell’ Università 6, 26900 Lodi, Italy;
| | - Giuseppe Sarli
- Department of Veterinary Medical Sciences, University of Bologna, Via Tolara di Sopra, 50, Ozzano dell’Emilia, 40064 Bologna, Italy; (G.S.); (C.B.)
| | - Cinzia Benazzi
- Department of Veterinary Medical Sciences, University of Bologna, Via Tolara di Sopra, 50, Ozzano dell’Emilia, 40064 Bologna, Italy; (G.S.); (C.B.)
| | - Marco Luigi Bianchi
- DOCVET Clinica Veterinaria Nervianese, Via Rho 2, 20014 Nerviano, Italy; (F.M.); (M.L.B.)
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14
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Haughey AM, Moloney BM, O'Brien CM. Epithelioid Haemangioendothelioma; Not simply a hepatic pathology. Clin Imaging 2023; 102:42-52. [PMID: 37541086 DOI: 10.1016/j.clinimag.2023.07.003] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/08/2023] [Revised: 06/26/2023] [Accepted: 07/16/2023] [Indexed: 08/06/2023]
Affiliation(s)
- Aoife M Haughey
- Department of Medical Imaging, JDMI University Health Network, University of Toronto, 610 University Avenue, Toronto, Ontario M5G 2M9, Canada
| | - Brian M Moloney
- The Christie NHS Foundation Trust, Wilmslow Rd, Manchester M20 4BX, United Kingdom
| | - Ciara M O'Brien
- Department of Medical Imaging, JDMI University Health Network, University of Toronto, 610 University Avenue, Toronto, Ontario M5G 2M9, Canada. ciara.o'
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15
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Gholami R, Soltani M, Salarieh N, Ketabi Moghadam P. A Liver-Derived Vascular Lesion: Hepatic Hemangioma or Hepatic Epithelioid Hemangioendothelioma? Middle East J Dig Dis 2023; 15:210-213. [PMID: 38023470 PMCID: PMC10660322 DOI: 10.34172/mejdd.2023.347] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/12/2023] [Accepted: 06/17/2023] [Indexed: 12/01/2023] Open
Abstract
Hepatic epithelioid hemangioendothelioma (EHE) is an uncommon vascular endothelial cell tumor of the liver with numerous symptoms and features. The median affected age is 41, and females are more frequently affected than men. In the following article, a 37-year-old nurse is presented who was referred to the hospital with severe right upper quadrant pain. She had been misdiagnosed with hepatic hemangioma for years, which finally turned out to be hepatic EHE. Liver transplantation has been recognized as the therapeutic method of choice due to the considerable extent of liver involvement and nonresponse to medications.
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Affiliation(s)
- Roham Gholami
- Fellowship of Gastroenterology, Department of Internal Medicine, School of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran
| | - Moslem Soltani
- Department of Internal Medicine, Islamic Azad University of Mashhad, Mashhad, Iran
| | - Naghmeh Salarieh
- Gastroenterologist and Hepatologist, Guilan University of Medical Sciences, Guilan, Iran
| | - Pardis Ketabi Moghadam
- Research Institute for Gastroenterology and Liver Diseases, Shahid Beheshti University of Medical Sciences, Tehran, Iran
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16
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Lieu DQ, Anh TN, Luan DT, Quynh MT, Duc NM. A rare case of hepatic epitheliod hemangioendothelioma. Radiol Case Rep 2023; 18:1695-1699. [PMID: 36895898 PMCID: PMC9989282 DOI: 10.1016/j.radcr.2023.01.106] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/21/2023] [Revised: 01/25/2023] [Accepted: 01/30/2023] [Indexed: 03/03/2023] Open
Abstract
Hepatic epithelioid hemangioendothelioma (HEHE) is an extremely uncommon tumor of the liver. It typically lacks recognizable clinical signs and is diagnosed with the aid of imaging and histopathology combined with immunohistochemical analysis. We discuss the case of a 40-year-old woman with HEHE. The aim of this case report and literature review is to increase doctors' knowledge of HEHE and reduce the incidence of missed clinical diagnoses.
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Affiliation(s)
- Dau Quang Lieu
- Department of Internal Medicine, Hanoi Medical University Hospital, Hanoi, Vietnam
| | - Tran Ngoc Anh
- Department of Internal Medicine, Hanoi Medical University Hospital, Hanoi, Vietnam
| | - Dao-Thi Luan
- Department of Pathology, Hanoi Medical University Hospital, Hanoi, Vietnam
| | - Mai-Thi Quynh
- Department of Radiology, Hanoi Medical University Hospital, Hanoi, Vietnam
| | - Nguyen Minh Duc
- Department of Radiology, Pham Ngoc Thach University of Medicine, 2 Duong Quang Trung Ward 12 District 10, Ho Chi Minh City, Vietnam
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17
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Chu K, Li Z, Tang W, Jiang X. Updated information regarding management of hepatic epithelioid hemangioendothelioma. Intractable Rare Dis Res 2022; 11:211-214. [PMID: 36457586 PMCID: PMC9709622 DOI: 10.5582/irdr.2022.01113] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/12/2022] [Revised: 11/04/2022] [Accepted: 11/06/2022] [Indexed: 11/10/2022] Open
Abstract
Hepatic epithelioid hemangioendothelioma (HEHE) is a rare hepatic vascular tumor with a borderline biological behavior between hemangioma and hemangiosarcoma. It tends to be multiple or diffuse subcapsular lesions across the liver but has no characteristic clinical manifestations or imaging findings. On computed tomography and magnetic resonance imaging, these lesions usually have a hypodense appearance with heterogeneous enhancement and a "halo sign" or "lollipop sign" may be evident in some cases. HEHE is diagnosed mainly based on a pathological examination along with differential immunohistochemical markers such as CAMTA1, CD31, CD34, CD10, vimentin, and factor VIII antigen. Currently, there are no standardized treatment guidelines for HEHE, and surgery (curative resection and liver transplantation) remains the mainstay of treatment. Studies have indicated that extra-hepatic metastasis might not be a contraindication for resection or transplantation. Systemic chemotherapeutic agents including doxorubicin, vincristine, interferon-a, 5-fluorouracil, and thalidomide, as well as VEGF-related agents are being investigated, but no agents have been approved for the treatment of HEHE.
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Affiliation(s)
- Kaijian Chu
- Biliary Surgery Department No. 1, Eastern Hepatobiliary Surgical Hospital, Naval Medical University, Shanghai, China
- Hepato-Biliary-Pancreatic Surgery Division, Department of Surgery, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Zhizheng Li
- Biliary Surgery Department No. 1, Eastern Hepatobiliary Surgical Hospital, Naval Medical University, Shanghai, China
| | - Wei Tang
- Hepato-Biliary-Pancreatic Surgery Division, Department of Surgery, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
- International Health Care Center, National Center for Global Health and Medicine, Tokyo, Japan
| | - Xiaoqing Jiang
- Biliary Surgery Department No. 1, Eastern Hepatobiliary Surgical Hospital, Naval Medical University, Shanghai, China
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18
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Kitsugi K, Kawata K, Matsumoto M, Umemura M, Hanaoka T, Yamashita M, Takatori S, Ito J, Ohta K, Chida T, Noritake H, Suda T. A Case of Hepatic Angiosarcoma with Peliosis Hepatis. Intern Med 2022; 62:1157-1166. [PMID: 36070948 PMCID: PMC10183276 DOI: 10.2169/internalmedicine.0315-22] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/09/2022] Open
Abstract
A 59-year-old woman presented to our hospital with liver dysfunction. Imaging revealed multiple lesions in the liver. The patient was diagnosed with peliosis hepatis using percutaneous and laparoscopic biopsies. However, her condition worsened with the appearance of new, obvious mass-forming lesions. Therefore, she underwent a second percutaneous biopsy of these lesions and was diagnosed with hepatic angiosarcoma. Her condition progressed rapidly, and she died two weeks after the diagnosis. Diagnosis of hepatic angiosarcoma in the early stages is difficult. It should be noted that hepatic angiosarcoma may be associated with the development of peliosis hepatis.
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Affiliation(s)
- Kensuke Kitsugi
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Kazuhito Kawata
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Moe Matsumoto
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Masahiro Umemura
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Tomohiko Hanaoka
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Maho Yamashita
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Shingo Takatori
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Jun Ito
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Kazuyoshi Ohta
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Takeshi Chida
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Hidenao Noritake
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
| | - Takafumi Suda
- Department of Internal Medicine II, Hamamatsu University School of Medicine, Japan
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19
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Shenoy A, Parikh ND. CAQ Corner: Liver transplantation for rare hepatobiliary malignancies. Liver Transpl 2022; 29:644-652. [PMID: 35726389 DOI: 10.1002/lt.26533] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/28/2022] [Revised: 05/12/2022] [Accepted: 05/26/2022] [Indexed: 01/12/2023]
Affiliation(s)
- Abhishek Shenoy
- Division of Gastroenterology and Hepatology, University of Michigan, Ann Arbor, Michigan, USA
| | - Neehar D Parikh
- Division of Gastroenterology and Hepatology, University of Michigan, Ann Arbor, Michigan, USA
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20
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A rare hepatic epithelioid hemangioendothelioma mimicking hepatocellular carcinoma in a patient with hepatitis. Am J Med Sci 2022; 364:e23-e24. [DOI: 10.1016/j.amjms.2022.03.012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/14/2021] [Accepted: 03/30/2022] [Indexed: 11/18/2022]
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21
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Kolleri JJ, Khaliq A, Ladumor SB, Habtezghi AB, Koshy SM, Petkar M. Primary Hepatic Epithelioid Hemangioendothelioma Masquerading as a Hepatic Abscess With Infective Picture: A Case Report. Cureus 2022; 14:e22859. [PMID: 35392441 PMCID: PMC8977811 DOI: 10.7759/cureus.22859] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 02/28/2022] [Indexed: 11/09/2022] Open
Abstract
Hepatic endotheiloid hemangioendothelioma (EHE) commonly presents with multilobar involvement with locally aggressive behavior. In most cases, it presents with right upper quadrant abdominal pain, hepatomegaly, and weight loss with metastasis commonly to the lung. We present a 33-year-old woman with hepatic EHE with an initial presentation mimicking hepatic abscess and imaging findings misleading for metastatic liver lesions. It was confirmed on pathology with immunohistochemistry, but the patient could not survive due to her late presentation and the presence of metastatic lesions in the lung.
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22
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Zhang W, Zhang H, Zhong Y, Zhang K, Kong H, Yu L, Chen Y, Bai Y, Zhu Z, Yang Y, Gao X. Novel and Specific MRI Features Indicate the Clinical Features of Patients With Rare Hepatic Tumor Epithelioid Hemangioendothelioma. Front Oncol 2022; 12:729177. [PMID: 35155220 PMCID: PMC8828502 DOI: 10.3389/fonc.2022.729177] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/23/2021] [Accepted: 01/03/2022] [Indexed: 12/11/2022] Open
Abstract
Objective To investigate the MRI features and clinical significance of hepatic epithelioid hemangioendothelioma (HEHE). Methods Clinical records and MRI findings were retrospectively evaluated in nine HEHE patients from May 2010 to January 2020. Result There were 121 lesions in nine patients with a predominantly peripheral distribution. Five lesions (4.13%) in two patients (22.22%) had evidence of capsular retraction, and three patients had lung metastasis (33.33%). Dynamic contrast-enhanced MRI showed progressive enhancement, mainly in two ways: ring enhancement with hypovascularity in four patients (44.44%) and ring enhancement with hypervascularity in five patients (55.56%). Imaging demonstrated a multilayer ring appearance, which was typically observed on T2-weighted imaging (T2WI). The most common appearance consisted of two layers of varying signal, with some images displaying up to four layers. There were significant differences in the size of lesions between different layers of multilayer ring appearance (p < 0.001). All lesions exhibited a two-layer appearance on diffusion-weighted imaging (DWI), with hyperintensity at the periphery and a slightly high signal at the center (except for those with a single layer on T2WI). The “vascular penetration sign” was observed in most lesions, and the blood vessels of 112 lesions (92.56%) were portal vein branches, and five (4.13%) were hepatic vein branches. Pulmonary metastasis was found in three patients with the “vascular penetration sign” of hepatic vein branches. Conclusion The multilayer ring appearance on T2WI, the “vascular penetration sign”, and the two enhancement patterns may be of great significance in the diagnosis and treatment of HEHE. The “vascular penetration sign” of hepatic vein branches may indicate extrahepatic metastasis.
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Affiliation(s)
- Wei Zhang
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
| | - Hongtao Zhang
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
| | - Yanwei Zhong
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
| | - Keming Zhang
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China.,Department of Hepatobiliary Surgery, Peking University International Hospital, Beijing, China
| | - Huifang Kong
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
| | - Linxiang Yu
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
| | - Yan Chen
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
| | - Yili Bai
- Beijing Shijitan Hospital, Capital Medical University, Beijing, China
| | - Zhenyu Zhu
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
| | - Yongping Yang
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
| | - Xudong Gao
- Department of Hepatology and Department of Radiology, The 5th Medical Center of PLA General Hospital, Beijing, China
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23
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Wang CI, Chu PM, Chen YL, Lin YH, Chen CY. Chemotherapeutic Drug-Regulated Cytokines Might Influence Therapeutic Efficacy in HCC. Int J Mol Sci 2021; 22:ijms222413627. [PMID: 34948424 PMCID: PMC8707970 DOI: 10.3390/ijms222413627] [Citation(s) in RCA: 22] [Impact Index Per Article: 5.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/26/2021] [Revised: 12/04/2021] [Accepted: 12/16/2021] [Indexed: 12/12/2022] Open
Abstract
Hepatocellular carcinoma (HCC), the most common type of liver cancer, is the second leading cause of cancer-related mortality worldwide. Processes involved in HCC progression and development, including cell transformation, proliferation, metastasis, and angiogenesis, are inflammation-associated carcinogenic processes because most cases of HCC develop from chronic liver damage and inflammation. Inflammation has been demonstrated to be a crucial factor inducing tumor development in various cancers, including HCC. Cytokines play critical roles in inflammation to accelerate tumor invasion and metastasis by mediating the migration of immune cells into damaged tissues in response to proinflammatory stimuli. Currently, surgical resection followed by chemotherapy is the most common curative therapeutic regimen for HCC. However, after chemotherapy, drug resistance is clearly observed, and cytokine secretion is dysregulated. Various chemotherapeutic agents, including cisplatin, etoposide, and 5-fluorouracil, demonstrate even lower efficacy in HCC than in other cancers. Tumor resistance to chemotherapeutic drugs is the key limitation of curative treatment and is responsible for treatment failure and recurrence, thus limiting the ability to treat patients with advanced HCC. Therefore, the capability to counteract drug resistance would be a major clinical advancement. In this review, we provide an overview of links between chemotherapeutic agents and inflammatory cytokine secretion in HCC. These links might provide insight into overcoming inflammatory reactions and cytokine secretion, ultimately counteracting chemotherapeutic resistance.
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Affiliation(s)
- Chun-I Wang
- Radiation Biology Research Center, Institute for Radiological Research, Chang Gung Memorial Hospital, Chang Gung University, Taoyuan 333, Taiwan;
| | - Pei-Ming Chu
- Department of Anatomy, School of Medicine, China Medical University, Taichung 404, Taiwan;
| | - Yi-Li Chen
- Department of Cell Biology and Anatomy, College of Medicine, National Cheng Kung University, Tainan 70101, Taiwan;
| | - Yang-Hsiang Lin
- Liver Research Center, Chang Gung Memorial Hospital, Linkou, Taoyuan 333, Taiwan;
| | - Cheng-Yi Chen
- Department of Cell Biology and Anatomy, College of Medicine, National Cheng Kung University, Tainan 70101, Taiwan;
- Correspondence: ; Tel./Fax: +886-6-2353535 (ext. 5329)
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24
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Serji B, Ramdani A, Mirali H, Bouhout T, Bennani A, El Harroudi T. Hepatic epithelioid hemangioendothelioma simulating liver metastasis: A case report. Ann Med Surg (Lond) 2021; 70:102885. [PMID: 34691426 PMCID: PMC8519773 DOI: 10.1016/j.amsu.2021.102885] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/26/2021] [Revised: 09/20/2021] [Accepted: 09/21/2021] [Indexed: 12/02/2022] Open
Abstract
Introduction Hepatic epithelioid hemangioendothelioma (HEHE) is a rare tumor of vascular origin. Liver involvement is often multinodular simulating metastases. Herein, we report a rare case of HEHE mimicking liver metastases in a female patient. Case presentation A 43-years-old female patient, presented with complaints of pain in the right upper quadrant. Laboratory tests were all within the normal range. Abdominal ultrasound revealed multiple heterogeneous hypoechoic liver lesions. A thoracic and abdominopelvic computed tomography showed bilateral pulmonary micronodules with multiple hypodense hepatic nodules involving both lobes. Percutaneous ultrasound-guided biopsy with pathological study and immunohistochemistry staining revealed the diagnosis of hepatic epithelioid hemangioendothelioma. Discussion HEHE usually involves both liver lobes with three radiological presentations: single nodular, multiple nodular, or diffuse types, the diffuse type reflects an advanced stage. 3/4 of the cases are initially misdiagnosed as liver metastases or primary liver tumors. The pathological study with the immunochemistry stainings confirms the diagnosis. There is no standard treatment for HEHE due to its rarity and lack of prospective randomized studies. Conclusion HEHE is a rare tumor of vascular origin of unknown etiology with malignant potential and unpredictable course. The therapeutic management of this rare condition is not codified and is discussed on a case-by-case basis. Surgical treatment remains the best option with an excellent outcome.
Hepatic epithelioid hemangioendothelioma is a rare tumor of vascular origin. The Clinical presentation and the radiological findings are not specific. Final Diagnosis is made upon pathological study and immunohistochemistry staining. Surgical treatment remain the treatment of choice.
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Affiliation(s)
- Badr Serji
- Surgical Oncology Department, Mohammed VI University Hospital, Regional Oncology Center, Oujda, Morocco.,Mohammed First University Oujda, Faculty of Medicine and Pharmacy Oujda, Oujda, Morocco
| | - Abdelbassir Ramdani
- Surgical Oncology Department, Mohammed VI University Hospital, Regional Oncology Center, Oujda, Morocco.,Mohammed First University Oujda, Faculty of Medicine and Pharmacy Oujda, Oujda, Morocco
| | - Houda Mirali
- Mohammed First University Oujda, Faculty of Medicine and Pharmacy Oujda, Oujda, Morocco.,Radiology Department, Mohammed VI University Hospital, Oujda, Morocco
| | - Tariq Bouhout
- Surgical Oncology Department, Mohammed VI University Hospital, Regional Oncology Center, Oujda, Morocco.,Mohammed First University Oujda, Faculty of Medicine and Pharmacy Oujda, Oujda, Morocco
| | - Amal Bennani
- Mohammed First University Oujda, Faculty of Medicine and Pharmacy Oujda, Oujda, Morocco.,Pathology Department, Mohammed VI University Hospital, Oujda, Morocco
| | - Tijani El Harroudi
- Surgical Oncology Department, Mohammed VI University Hospital, Regional Oncology Center, Oujda, Morocco.,Mohammed First University Oujda, Faculty of Medicine and Pharmacy Oujda, Oujda, Morocco
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25
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Ostojic A, Mrzljak A, Mikulic D. Liver transplantation for benign liver tumors. World J Hepatol 2021; 13:1098-1106. [PMID: 34630877 PMCID: PMC8473500 DOI: 10.4254/wjh.v13.i9.1098] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/24/2021] [Revised: 05/12/2021] [Accepted: 07/21/2021] [Indexed: 02/06/2023] Open
Abstract
Benign liver tumors are common lesions that are usually asymptomatic and are often found incidentally due to recent advances in imaging techniques and their widespread use. Although most of these tumors can be managed conservatively or treated by surgical resection, liver transplantation (LT) is the only treatment option in selected patients. LT is usually indicated in patients that present with life-threatening complications, when the lesions are diffuse in the hepatic parenchyma or when malignant transformation cannot be ruled out. However, due to the significant postoperative morbidity of the procedure, scarcity of available donor liver grafts, and the benign course of the disease, the indications for LT are still not standardized. Hepatic adenoma and adenomatosis, hepatic hemangioma, and hepatic epithelioid hemangioendothelioma are among the most common benign liver tumors treated by LT. This article reviews the role of LT in patients with benign liver tumors. The indications for LT and long-term outcomes of LT are presented.
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Affiliation(s)
- Ana Ostojic
- Department of Gastroenterology and Hepatology, University Hospital Center Zagreb, Zagreb 10000, Croatia
| | - Anna Mrzljak
- Department of Gastroenterology and Hepatology, University Hospital Center Zagreb, Zagreb 10000, Croatia
| | - Danko Mikulic
- Department of Surgery, University Hospital Merkur, Zagreb 10000, Croatia.
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26
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Talakić E, Janek E, Mikalauskas S, Schemmer P. Liver Transplantation in Malignancies: A Comprehensive and Systematic Review on Oncological Outcome. Visc Med 2021; 37:302-314. [PMID: 34540947 PMCID: PMC8406343 DOI: 10.1159/000517328] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/02/2021] [Accepted: 05/20/2021] [Indexed: 12/13/2022] Open
Abstract
INTRODUCTION Liver transplantation (LT) is today's standard treatment for both end-stage liver disease and tumors; however, suitable grafts for LT are a scarce resource and outcome after LT is highly dependent on its underlying indication. Thus, patients must be carefully selected to optimize the number of life years gained per graft. This comprehensive and systematic review critically reflects the most recently published oncological outcome data after LT in malignancies based on the preoperative radiological findings. METHODS A systematic literature search was conducted to detect preferentially most recent high-volume series or large database analysis on oncological outcomes after LT for both primary liver cancer and liver metastases between January 1, 2019, and November 14, 2020. A comprehensive review on the radiological assessment of the reviewed liver malignancies is included and its preoperative value for an outcome-driven indication reflected. RESULTS Twenty most recent high-volume or relevant studies including a total number of 2,521 patients were identified including 4, 4, 4, 4, 3, and 1 publications on oncological outcome after LT for hepatocellular carcinoma, cholangiocellular carcinoma, hepatic epitheloid hemangioendothelioma, hepatoblastoma, and both metastatic neuroendocrine tumors and colorectal cancer, respectively. The overall survival is comparable to patients without tumors if patients with malignancies are well selected for LT; however, this is highly dependent on tumor entity, tumor stage, and both neoadjuvant and concomitant treatment. DISCUSSION/CONCLUSION LT is a promising option for better survival in patients with malignant liver tumors in selected patients; however, the indication must be critically discussed prior to LT in every single case in the context of organ shortage.
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Affiliation(s)
- Emina Talakić
- Division of General Radiology, Department of Radiology, Medical University Graz (MUG), Graz, Austria
- Transplant Center Graz, Medical University Graz (MUG), Graz, Austria
| | - Elmar Janek
- Division of General Radiology, Department of Radiology, Medical University Graz (MUG), Graz, Austria
- Transplant Center Graz, Medical University Graz (MUG), Graz, Austria
| | - Saulius Mikalauskas
- Transplant Center Graz, Medical University Graz (MUG), Graz, Austria
- General, Visceral and Transplant Surgery, Department of Surgery, Medical University Graz (MUG), Graz, Austria
| | - Peter Schemmer
- Transplant Center Graz, Medical University Graz (MUG), Graz, Austria
- General, Visceral and Transplant Surgery, Department of Surgery, Medical University Graz (MUG), Graz, Austria
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27
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Hepatic Epithelioid Hemangioendothelioma: A Review of Three Liver Transplant Recipients. HEPATITIS MONTHLY 2021. [DOI: 10.5812/hepatmon.114580] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 02/13/2023]
Abstract
Introduction: Hepatic epithelioid hemangioendothelioma (HEHE) is a discrete vascular tumor with an unpredictable natural course. This rare tumor is commonly found incidentally and not too often is mistaken radiologically and histologically for another tumor. No single treatment strategy has yet been established for it, partly due to its variable clinical course, ranging from an indolent tumor with prolonged survival to an aggressive, fatal disease. Case Presentation: Among 1,029 liver transplantation cases performed at our hospital between January 2001 and November 2019, three were done for HEHE. In this study, we present these three cases and review their clinical and histopathologic characteristics. Conclusions: Epithelioid hemangioendothelioma (EHE) of the liver is a low-grade malignant tumor with different presentations, treatment strategies, and outcomes. The histopathologic characteristics of HEHE can hide its vascular nature, thus leading to its confusion with other lesions. This tumor is unique in that its clinical and histopathologic features do not always correlate with its biologic behavior. There are no reliable criteria in predicting the clinical outcome of HEHE, which needs further research.
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Chahrour MA, Khachfe HH, Habib JR, El-Asmar R, Saifi O, Jamali FR. Treatment and Prognosis of Hepatic Epithelioid Hemangioendothelioma: A SEER Database Analysis. World J Surg 2021; 45:2886-2894. [PMID: 33999226 DOI: 10.1007/s00268-021-06165-6] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 04/24/2021] [Indexed: 12/20/2022]
Abstract
BACKGROUND Hepatic epithelioid hemangioendothelioma (HEH) is a rare tumor that can affect multiple organs. Little is known about the pathophysiology, clinical course and management of this disease. The aim of this study is to determine survival rates and elucidate the role of various prognostic factors and therapeutic modalities as compared to surgery on patients with HEH. METHODS A retrospective analysis on patients diagnosed with HEH between 2004 and 2016 was performed utilizing the SEER database. Kaplan-Meier curves were constructed to determine overall and cancer-specific survival, and the log-rank test was used to compare between groups. To explore prognostic factors and treatment outcomes, univariable and multivariable Cox proportional hazard models were developed. RESULTS A total of 353 patients with HEH (median age: 50.4 years) were identified. The most common surgery performed was liver resection (90.8%). One-year OS in the surgical group and non-surgical group was 86.6% and 61.0%, respectively, while 5-year OS was 75.2% and 37.4%, respectively. On multivariable analysis, surgery emerged as a favorable prognostic factor [HR (95%CI): 0.404 (0.215-0.758) p value = 0.005]. Age > 65 years [HR (95%CI): 2.548 (1.442-4.506) p value = 0.001] and tumor size > 10 cm [HR (95%CI): 2.401 (1.319-4.37) p value = 0.004] were shown to be poor survival prognostic factors. CONCLUSION HEH is a rare disease that is poorly understood. Surgical intervention is associated with improved survival rates. Multicenter prospective collaborations are needed to improve our limited knowledge about this neoplasm and determine the optimal treatment strategy.
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Affiliation(s)
- Mohamad A Chahrour
- Department of Surgery, American University of Beirut Medical Center, Beirut, Lebanon
| | - Hussein H Khachfe
- Department of Surgery, American University of Beirut Medical Center, Beirut, Lebanon
| | - Joseph R Habib
- Department of Surgery, Johns Hopkins University, Baltimore, MD, USA
| | - Rudy El-Asmar
- Department of Surgery, American University of Beirut Medical Center, Beirut, Lebanon
| | - Omran Saifi
- Department of Radiation Oncology, Mayo Clinic, Jacksonville, FL, USA
| | - Faek R Jamali
- Division of General Surgery, Sheikh Shakhbout Medical City, Abu Dhabi, United Arab Emirates.
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Gigante E, Paradis V, Ronot M, Cauchy F, Soubrane O, Ganne-Carrié N, Nault JC. New insights into the pathophysiology and clinical care of rare primary liver cancers. JHEP Rep 2021; 3:100174. [PMID: 33205035 PMCID: PMC7653076 DOI: 10.1016/j.jhepr.2020.100174] [Citation(s) in RCA: 34] [Impact Index Per Article: 8.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/04/2020] [Revised: 08/06/2020] [Accepted: 08/10/2020] [Indexed: 02/07/2023] Open
Abstract
Hepatocholangiocarcinoma, fibrolamellar carcinoma, hepatic haemangioendothelioma and hepatic angiosarcoma represent less than 5% of primary liver cancers. Fibrolamellar carcinoma and hepatic haemangioendothelioma are driven by unique somatic genetic alterations (DNAJB1-PRKCA and CAMTA1-WWTR1 fusions, respectively), while the pathogenesis of hepatocholangiocarcinoma remains more complex, as suggested by its histological diversity. Histology is the gold standard for diagnosis, which remains challenging even in an expert centre because of the low incidences of these liver cancers. Resection, when feasible, is the cornerstone of treatment, together with liver transplantation for hepatic haemangioendothelioma. The role of locoregional therapies and systemic treatments remains poorly studied. In this review, we aim to describe the recent advances in terms of diagnosis and clinical management of these rare primary liver cancers.
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Key Words
- 5-FU, 5-Fluorouracil
- AFP, alpha-fetoprotein
- APHE, arterial phase hyperenhancement
- CA19-9, carbohydrate antigen 19-9
- CCA, cholangiocarcinoma
- CEUS, contrast-enhanced ultrasound
- CK, cytokeratin
- CLC, cholangiolocellular carcinoma
- EpCAM, epithelial cell adhesion molecule
- FISH, fluorescence in situ hybridisation
- FLC, fibrolamellar carcinoma
- Fibrolamellar carcinoma
- HAS, hepatic angiosarcoma
- HCC, hepatocellular carcinoma
- HEH, hepatic epithelioid haemangioendothelioma
- HepPar1, hepatocyte specific antigen antibody
- Hepatic angiosarcoma
- Hepatic hemangioendothelioma
- Hepatocellular carcinoma
- Hepatocholangiocarcinoma
- IHC, immunohistochemistry
- LI-RADS, liver imaging reporting and data system
- LT, liver transplantation
- Mixed tumor
- RT-PCR, reverse transcription PCR
- SIRT, selective internal radiation therapy
- TACE, transarterial chemoembolisation
- WHO, World Health Organization
- cHCC-CCA, combined hepatocholangiocarcinoma
- iCCA, intrahepatic cholangiocarcinoma
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Affiliation(s)
- Elia Gigante
- Service d’hépatologie, Hôpital Avicenne, Hôpitaux Universitaires Paris-Seine-Saint-Denis, Assistance-Publique Hôpitaux de Paris, Bobigny, France
- Centre de recherche sur l’inflammation, Inserm, Université de Paris, INSERM UMR 1149 « De l'inflammation au cancer », Paris, France
- Unité de Formation et de Recherche Santé Médecine et Biologie Humaine, Université Paris 13, Paris, France
| | - Valérie Paradis
- Centre de recherche sur l’inflammation, Inserm, Université de Paris, INSERM UMR 1149 « De l'inflammation au cancer », Paris, France
- Service d'anatomie pathologique, Hôpitaux Universitaires Paris-Nord-Val-de-Seine, Assistance-Publique Hôpitaux de Paris, Clichy, France
- Université de Paris, Paris, France
| | - Maxime Ronot
- Centre de recherche sur l’inflammation, Inserm, Université de Paris, INSERM UMR 1149 « De l'inflammation au cancer », Paris, France
- Service de radiologie, Hôpital Beaujon, Hôpitaux Universitaires Paris-Nord-Val-de-Seine, Assistance-Publique Hôpitaux de Paris, Clichy, France
- Université de Paris, Paris, France
| | - François Cauchy
- Centre de recherche sur l’inflammation, Inserm, Université de Paris, INSERM UMR 1149 « De l'inflammation au cancer », Paris, France
- Service de chirurgie hépato-bilio-pancréatique et transplantation hépatique, Hôpitaux Universitaires Paris-Nord-Val-de-Seine, Assistance-Publique Hôpitaux de Paris, Clichy, France
- Université de Paris, Paris, France
| | - Olivier Soubrane
- Centre de recherche sur l’inflammation, Inserm, Université de Paris, INSERM UMR 1149 « De l'inflammation au cancer », Paris, France
- Service de chirurgie hépato-bilio-pancréatique et transplantation hépatique, Hôpitaux Universitaires Paris-Nord-Val-de-Seine, Assistance-Publique Hôpitaux de Paris, Clichy, France
- Université de Paris, Paris, France
| | - Nathalie Ganne-Carrié
- Service d’hépatologie, Hôpital Avicenne, Hôpitaux Universitaires Paris-Seine-Saint-Denis, Assistance-Publique Hôpitaux de Paris, Bobigny, France
- Unité de Formation et de Recherche Santé Médecine et Biologie Humaine, Université Paris 13, Paris, France
- Centre de Recherche des Cordeliers, Inserm, Sorbonne Université, Université Paris, INSERM UMR 1138, Functional Genomics of Solid Tumors, F-75006, Paris, France
| | - Jean-Charles Nault
- Service d’hépatologie, Hôpital Avicenne, Hôpitaux Universitaires Paris-Seine-Saint-Denis, Assistance-Publique Hôpitaux de Paris, Bobigny, France
- Unité de Formation et de Recherche Santé Médecine et Biologie Humaine, Université Paris 13, Paris, France
- Centre de Recherche des Cordeliers, Inserm, Sorbonne Université, Université Paris, INSERM UMR 1138, Functional Genomics of Solid Tumors, F-75006, Paris, France
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Cifrián Canales I, García Bernardo CM, Contreras Saiz E. Hepatic epithelioid hemangioendothelioma: a rare vascular tumor with an unpredictable diagnosis. REVISTA ESPANOLA DE ENFERMEDADES DIGESTIVAS 2020; 113:300-301. [PMID: 33222478 DOI: 10.17235/reed.2020.7318/2020] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
We present the case of a 58-year-old female with no relevant medical or surgical history, who was referred to our hospital with a solitary hepatic lesion. She presented a slightly abnormal liver function in a routine blood test: ALT 71 U/l (range 0-33), AST 40 U/l (range 0-32) and GGT 71 U/l (range 0-40), with no symptoms. Ultrasound imaging showed a 3-cm-size focal lesion with a peripheral hypoechoic rim in the right hepatic lobe. Abdominal computed tomography (CT) scan after contrast injection revealed a subcapsular hypodense lesion in segment VII with capsular retraction. Other extrahepatic lesions were not seen.
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Xu F, Zheng J, Fu M, Zhou H. Antiprogrammed cell death protein 1 immunotherapy for angiosarcoma with high programmed death-ligand 1 expression: a case report. Immunotherapy 2020; 12:771-776. [PMID: 32611263 DOI: 10.2217/imt-2020-0122] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/24/2023] Open
Abstract
Background: Angiosarcoma (AS) is a rare malignancy originating from lymphatic or vascular endothelial cells. Prognosis of the disease is usually dismal and there is no effective treatment. Immunotherapy has been proved to be effective for various cancer types. Programmed death-ligand 1 (PD-L1) expression is generally recognized as a biomarker for the prediction of response to anti-PD-(L)1 immunotherapies. Methods & results: Here, we discuss a single case by highlighting the treatment of the antiprogrammed cell death protein 1 drug pembrolizumab with high PD-L1 expression. CT scan demonstrated a confirmed size reduction of some lesions compared with original lesions, which indicates the possible clinical benefit. Conclusion: We speculate that early anti-PD-1 treatment may be a promising strategy for angiosarcoma patients with high PD-L1 expression.
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Affiliation(s)
- Fei Xu
- Department of Respiratory Diseases, The First Affiliated Hospital of College of Medicine, Zhejiang University, Hangzhou, China
| | - Jing Zheng
- Department of Respiratory Diseases, The First Affiliated Hospital of College of Medicine, Zhejiang University, Hangzhou, China
| | - Mengjiao Fu
- Department of Respiratory Diseases, The First Affiliated Hospital of College of Medicine, Zhejiang University, Hangzhou, China
| | - Hua Zhou
- Department of Respiratory Diseases, The First Affiliated Hospital of College of Medicine, Zhejiang University, Hangzhou, China
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