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1
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Yang S, Tang Y, Yuan Z, Zhang J. Inflammatory myofibroblastic tumor in the liver after bone marrow transplantation: case report and literature review. Front Med (Lausanne) 2025; 12:1489399. [PMID: 40224630 PMCID: PMC11986994 DOI: 10.3389/fmed.2025.1489399] [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] [Received: 09/01/2024] [Accepted: 02/17/2025] [Indexed: 04/15/2025] Open
Abstract
Introduction Inflammatory myofibroblastic tumor (IMT) is a rare low-grade malignant neoplasm in the liver. Timely diagnosis and treatment of IMT are challenging due to its atypical symptoms and imaging results. Case report We report a 46-year-old woman who presented to our hospital with persistent hyperpyrexia and discomfort in the right upper abdomen for 2 months post bone marrow transplantation. Radiological findings revealed a space-occupying lesion of uncertain nature in the liver. Since the histological examination of the biopsy specimen indicated IMT, she underwent surgical resection. Subsequently, the postoperative pathology confirmed the diagnosis of IMT. The patient's febrile condition subsided after the surgery. A magnetic resonance imaging (MRI) scan performed 8 months later showed no signs of recurrence. Conclusion IMTs are caused by genetic rearrangements. Diagnosing IMT can be challenging especially in this case as we had to differentiate the tumor from inflammatory diseases associated with bone marrow transplantation. Hence, a thorough pathological immunohistochemical examination is required to confirm its diagnosis. Local IMTs should be treated with radical surgical resection. In cases of distant metastasis or incomplete resection cases, chemotherapy, targeted therapy, or immunotherapy can be utilized. Regular follow-up is crucial for improving the patient's survival rate.
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Affiliation(s)
| | | | | | - Jianwen Zhang
- Department of Hepatic Surgery and Liver Transplantation Center, Third Affiliated Hospital of Sun Yat-sen University, Guangzhou, China
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2
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Konstantinoff KS, Ludwig DR, Sharbidre K, Arif-Tiwari H, Itani M. Gallbladder and biliary pathology: lessons learned from multidisciplinary conference. Abdom Radiol (NY) 2025; 50:916-935. [PMID: 39259353 DOI: 10.1007/s00261-024-04533-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: 06/28/2024] [Revised: 08/12/2024] [Accepted: 08/14/2024] [Indexed: 09/13/2024]
Abstract
The radiologic diagnosis of biliary disease can be challenging due to atypical or delayed presentation, rare or less common entities, and imaging overlap of benign and malignant processes. Establishing a specific diagnosis, when possible, is important to avoid progression of infections to sepsis and multiorgan failure, and for appropriate staging and management in cases of malignancy. Gallstones are the most common biliary disease, and along with stone-related complications, including cholecystitis and choledocholithiasis, constitute the majority of acute biliary pathology. Late and atypical manifestations of acute cholecystitis demonstrate imaging overlap with primary gallbladder cholangiocarcinoma, especially with cases of exuberant inflammatory reaction such as xanthogranulomatous cholecystitis. Additional challenging scenarios related to gallbladder disease, that may be benign or malignant, include adjacent fistulas and lymphadenopathy. Dropped gallstones, especially in atypical locations, may be misdiagnosed as neoplastic. Recurrent cholecystitis after cholecystectomy, whether related to subtotal cholecystectomy or to stumpitis, is another entity that can be confusing to the radiologist with a documented history of cholecystectomy. Inflammatory and autoimmune conditions, such as pseudotumors and IgG4 disease, are a less common but not infrequent cause of diagnostic dilemma. Furthermore, biliary strictures and hepatobiliary cystic lesions can be benign or malignant and could constitute a diagnostic and management challenge. The goal of this manuscript is to present the lessons learned from multidisciplinary conferences on the above entities and suggest tips and pearls to maximize the value of radiologists' contribution to patient management.
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Affiliation(s)
- Katerina S Konstantinoff
- Mallinckrodt Institute of Radiology, Washington University in St. Louis, 510 S. Kingshighway Blvd, St., St. Louis, MO, 63110, USA
| | - Daniel R Ludwig
- Mallinckrodt Institute of Radiology, Washington University in St. Louis, 510 S. Kingshighway Blvd, St., St. Louis, MO, 63110, USA
| | - Kedar Sharbidre
- Department of Radiology, University of Alabama in Birmingham, 500 22nd Street South, Birmingham, AL, 35233, USA
| | - Hina Arif-Tiwari
- Department of Medical Imaging, University of Arizona College of Medicine, 1501 N. Campbell, Tucson, AZ, 85724, USA
| | - Malak Itani
- Mallinckrodt Institute of Radiology, Washington University in St. Louis, 510 S. Kingshighway Blvd, St., St. Louis, MO, 63110, USA.
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3
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Zengin A, Şendil AM, Angın YS, Türker B, Kılıç M, Ulaş M, Gündoğdu E, Arık D. Tumor in the liver: Six inflammatory pseudotumor patient. Turk J Surg 2024; 40:256-260. [PMID: 39917402 PMCID: PMC11792901 DOI: 10.47717/turkjsurg.2024.6479] [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: 06/12/2024] [Accepted: 09/18/2024] [Indexed: 01/05/2025]
Abstract
Inflammatory pseudotumor (IPT) is a rare liver disease confused with liver tumors. It is a disease that should be known in the differential diagnosis for clinicians as the correct diagnosis of IPT will prevent unnecessary surgery. Demographic datas, diagnoses and imaging modalities of six patients with radiologically and/or histopathologically diagnosed hepatic IPT between 2016 and 2023 were retrospectively analyzed. Four out of six patients were female and median age was 57.5 (47-66). C-reactive protein was higher in four patients, and carbohydrate antigen 19.9 level was higher in one patient. We used magnetic resonance imaging (MRI) for diagnosis in five patients. Only in one patient computed tomography was enough for diagnosis. Tumor locations were segment 5 for two patients, segment 7-8 in two patients, segment 7 in one patient, and 8 in one patient. Liver biopsy was performed in five patients because it could not be distinguished from malignancy by imaging methods. Histopathological results of all these biopsies defined as IPT. Initial tumor median size was 31 (17-55) mm. Two patients were operated on. The first one underwent right hepatectomy due to a 2-fold increase in size within 11 months. The second one had a mass indistinguishable from hepatic adenoma by MRI and underwent nonanatomic resection. In one patient, IPT disappeared completely in the 18th month of follow-up period while it regressed in size in two patients. Two of our patients had a history of recurrent endoscopic retrograde cholangiopancreatography, which we noticed incidentally before IPT was diagnosed. IPTs are liver masses with low malignant potential and may shrink spontaneously during follow-up. We suspected that biliary tract interventions may be the cause of IPT.
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Affiliation(s)
- Akile Zengin
- Department of Gastrointestinal Surgery, Eskişehir Osmangazi University Faculty of Medicine, Eskişehir, Türkiye
| | - Ahmet Murat Şendil
- Department of General Surgery, Eskişehir Osmangazi University Faculty of Medicine, Eskişehir, Türkiye
| | - Yavuz Selim Angın
- Department of General Surgery, Eskişehir Osmangazi University Faculty of Medicine, Eskişehir, Türkiye
| | - Barış Türker
- Clinic of Gastrointestinal Surgery, Çanakkale Mehmet Akif Ersoy State Hospital, Çanakkale, Türkiye
| | - Mehmet Kılıç
- Department of General Surgery, Eskişehir Osmangazi University Faculty of Medicine, Eskişehir, Türkiye
| | - Murat Ulaş
- Department of Gastrointestinal Surgery, Eskişehir Osmangazi University Faculty of Medicine, Eskişehir, Türkiye
| | - Elif Gündoğdu
- Department of Radiology, Eskişehir Osmangazi University Faculty of Medicine, Eskişehir, Türkiye
| | - Deniz Arık
- Department of Pathology, Eskişehir Osmangazi University Faculty of Medicine, Eskişehir, Türkiye
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4
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Desai G, Parikh DM, Wagle PK. A Giant Solid-Cystic Gastric Inflammatory Myofibroblastic Tumor: A Case Report and Literature Review. Cureus 2023; 15:e37167. [PMID: 37168160 PMCID: PMC10166389 DOI: 10.7759/cureus.37167] [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: 04/05/2023] [Indexed: 05/13/2023] Open
Abstract
An inflammatory myofibroblastic tumor (IMT) is a very rare tumor of mesenchymal origin with unclear etiopathogenesis, no unique diagnostic features, and no specific management protocol. It is often confused with inflammatory pseudotumor in literature, and the distinction needs further study. The average size, recurrence risk, and metastatic potential differ as per the site of origin. The abdomen is a very rare site for IMTs. Hepatic IMTs (H-IMTs) are reported to be solid tumors with sizes ranging from 1 cm to 20 cm in literature, and gastric IMTs (G-IMTs) range from 3 cm to 10 cm in size and can be solid-cystic. We report here a case of a 36-year-old gentleman with a 34x27x17 cm solid-cystic lesion in the lesser sac with loss of fat planes with stomach and left hemi-liver. The patient was managed by complete surgical resection of the lesion with wedge gastrectomy and wedge hepatectomy and recovered uneventfully. To our knowledge and based on our literature review, this case presents the largest reported and solid-cystic G-IMT with the involvement of left hemi-liver in a young gentleman and discusses its management as well as the relevant literature on this rare entity. This clinical presentation of G-IMT should be kept in the differential diagnosis in a relevant case presenting in the future. Immunohistochemistry is a must to establish the diagnosis, and surgical resection to negative margins is the management option of choice in resectable cases.
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Affiliation(s)
- Gunjan Desai
- Gastrointestinal Surgery, Asian Cancer Institute and ACI Cumballa Hill Hospital, Mumbai, IND
- Gastrointestinal Surgery, Lilavati Hospital and Research Centre, Mumbai, IND
| | - Deepak M Parikh
- Head and Neck Surgery, Asian Cancer Institute and ACI Cumballa Hill Hospital, Mumbai, IND
- Head and Neck Surgery, Lilavati Hospital and Research Centre, Mumbai, IND
| | - Prasad K Wagle
- Gastrointestinal Surgery, Asian Cancer Institute and ACI Cumballa Hill Hospital, Mumbai, IND
- Gastrointestinal Surgery, Lilavati Hospital and Research Centre, Mumbai, IND
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5
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Costa CM, Neto JS, Benavidez MR, Vincenzi R, Roda K, Oliveira CMV, Travassos NPR, de Lima LGCA, Pereira FP, Fonseca EA. Liver transplantation for hilar inflammatory myofibroblastic tumor: Case report and review of the literature. Pediatr Transplant 2022; 27:e14445. [PMID: 36435975 DOI: 10.1111/petr.14445] [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: 06/28/2022] [Revised: 09/15/2022] [Accepted: 11/15/2022] [Indexed: 11/28/2022]
Abstract
BACKGROUND Inflammatory myofibroblastic tumors (IMTs) of the liver are rare neoplasms. These tumors are difficult to distinguish from other neoplasms by radiological examination, have uncertain evolution, and there is no consensus on the treatment of these lesions. Hilar tumors can involve the portal vein, hepatic artery, bile duct, and spread to the branches of the portal triad, causing obstructive symptoms, occlusive phlebitis, and portal hypertension. Thus, liver transplantation (LT) is an excellent alternative for locally advanced hilar tumors, since it ensures complete tumor resection with free margins. METHODS/RESULTS We present a literature review and a case report showing a 3-year-old boy with liver IMT invading the hepatic hilum and inferior vena cava, who underwent a successful living donor liver transplantation that required portal vein and vena cava replacement. CONCLUSION The incidence of hilar IMTs is low and there is no well-established standard treatment. Liver transplantation for a hilar tumor with vascular invasion was acceptable in this case because the radically of the surgery was the key for the cure, and because the tumor was benign in nature.
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Affiliation(s)
- Carolina Magalhães Costa
- Hepatology and Liver Transplantation, Hospital Sírio-Libanês, São Paulo, Brazil.,Hepatology and Liver Transplantation, A. C. Camargo Cancer Center, São Paulo, Brazil
| | - João Seda Neto
- Hepatology and Liver Transplantation, Hospital Sírio-Libanês, São Paulo, Brazil.,Hepatology and Liver Transplantation, A. C. Camargo Cancer Center, São Paulo, Brazil
| | - Marcel R Benavidez
- Hepatology and Liver Transplantation, Hospital Sírio-Libanês, São Paulo, Brazil.,Hepatology and Liver Transplantation, A. C. Camargo Cancer Center, São Paulo, Brazil
| | - Rodrigo Vincenzi
- Hepatology and Liver Transplantation, Hospital Sírio-Libanês, São Paulo, Brazil.,Hepatology and Liver Transplantation, A. C. Camargo Cancer Center, São Paulo, Brazil
| | - Karina Roda
- Hepatology and Liver Transplantation, Hospital Sírio-Libanês, São Paulo, Brazil.,Hepatology and Liver Transplantation, A. C. Camargo Cancer Center, São Paulo, Brazil
| | - Caio Márcio V Oliveira
- Hepatology and Liver Transplantation, Hospital Sírio-Libanês, São Paulo, Brazil.,Hepatology and Liver Transplantation, A. C. Camargo Cancer Center, São Paulo, Brazil
| | - Nathália Porto R Travassos
- Hepatology and Liver Transplantation, Hospital Sírio-Libanês, São Paulo, Brazil.,Hepatology and Liver Transplantation, A. C. Camargo Cancer Center, São Paulo, Brazil
| | | | | | - Eduardo A Fonseca
- Hepatology and Liver Transplantation, Hospital Sírio-Libanês, São Paulo, Brazil.,Hepatology and Liver Transplantation, A. C. Camargo Cancer Center, São Paulo, Brazil
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6
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Gao SQ, Bao YJ, Luo JS. Inflammatory Myofibroblastic Tumor of the Hilar Bile Duct: A Case Report and Literature Review. Front Surg 2022; 9:928669. [PMID: 36211291 PMCID: PMC9537756 DOI: 10.3389/fsurg.2022.928669] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/26/2022] [Accepted: 06/03/2022] [Indexed: 11/13/2022] Open
Abstract
Background Inflammatory myofibroblastic tumor (IMT) is a very rare tumor and occurs seldom in the biliary tract. IMT can occur in any part of the body and in people of any age; however, it most commonly occurs in children or adolescents. Its etiology and pathogenesis are currently unknown. The clinical manifestations of a hilar inflammatory myofibroblastic tumor are atypical, and the imaging examination is nonspecific. The diagnosis is mainly based on histopathology and immunohistochemistry findings, and surgical resection is the preferred treatment method. Case Description Herein, we report a rare case of hilar bile duct IMT and review the related literature. Our patient was a 54-year-old woman presenting with a 1-day history of upper abdominal pain as the main clinical symptom. She was misdiagnosed as having cholangiocarcinoma before the surgery. She underwent surgery and was ultimately diagnosed with IMT based on histopathology and immunohistochemistry findings. On 1-year follow-up, no tumor recurrence or related complications were noted. Conclusions We hope this case report helps clinicians gain a deeper understanding of biliary IMT of the hilum.
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7
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Gros L, Dei Tos AP, Jones RL, Digklia A. Inflammatory Myofibroblastic Tumour: State of the Art. Cancers (Basel) 2022; 14:cancers14153662. [PMID: 35954326 PMCID: PMC9367282 DOI: 10.3390/cancers14153662] [Citation(s) in RCA: 31] [Impact Index Per Article: 10.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/24/2022] [Revised: 07/21/2022] [Accepted: 07/25/2022] [Indexed: 11/16/2022] Open
Abstract
Simple Summary Among sarcomas, which are rare cancers, inflammatory myofibroblastic tumors are extremely rare. Unlike other subtypes, this is a largely oncogene-driven neoplasia, and early gene rearrangement identification is important for accurate advanced stage treatment. In this manuscript, we review the clinicopathologic characteristics of this ultra-rare entity, as well as the current treatment landscape, with a particular focus on opportunities provided by tyrosine kinase inhibitors (TKIs). Abstract An inflammatory myofibroblastic tumor (IMT) is a neoplasm composed of myofibroblastic and fibroblastic spindle cells accompanied by inflammatory cells, including lymphocytes and eosinophils. It is an ultra-rare tumor, the optimal management of which remains to be defined. Surgery is the treatment of choice for localized tumors. The treatment of advanced disease is not precisely defined. Chemotherapy regimens result in an overall response rate of approximately 50% based on retrospective data. The latest pathophysiological data highlight the role played by tyrosine kinase fusion genes in IMT proliferation. Anaplast lymphoma kinase (ALK) oncogenic activation mechanisms have been characterized in approximately 80% of IMTs. In this context, data regarding targeted therapies are most important. The aims of this article are to review the latest published data on the use of systematic therapy, particularly the use of molecular targeted therapy, and to publish an additional case of an IMT with Ran-binding protein 2 (RANPB2)-ALK fusion showing a long response to a tyrosine kinase inhibitor.
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Affiliation(s)
- Louis Gros
- Department of Oncology, Lausanne University Hospital, University of Lausanne, 1011 Lausanne, Switzerland;
| | - Angelo Paolo Dei Tos
- Department of Pathology, Azienda Ospedale Università Padova, 35128 Padua, Italy;
- Department of Medicine, University of Padua School of Medicine, 35128 Padua, Italy
| | - Robin L. Jones
- Sarcoma Unit, The Royal Marsden NHS Foundation Trust, London SW3 6JJ, UK;
- Division of Clinical Sciences, Institute of Cancer Research, Royal Marsden Hospital, London SW3 6JJ, UK
| | - Antonia Digklia
- Department of Oncology, Lausanne University Hospital, University of Lausanne, 1011 Lausanne, Switzerland;
- Center of Sarcoma, Department of Oncology, Lausanne University Hospital, University of Lausanne, 1011 Lausanne, Switzerland
- Correspondence:
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8
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Rela M, Rammohan A, Reddy MS. Liver Tumors in Children. TEXTBOOK OF PEDIATRIC GASTROENTEROLOGY, HEPATOLOGY AND NUTRITION 2022:983-994. [DOI: 10.1007/978-3-030-80068-0_72] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/03/2025]
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9
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Strainiene S, Sedleckaite K, Jarasunas J, Savlan I, Stanaitis J, Stundiene I, Strainys T, Liakina V, Valantinas J. Complicated course of biliary inflammatory myofibroblastic tumor mimicking hilar cholangiocarcinoma: A case report and literature review. World J Clin Cases 2021; 9:6155-6169. [PMID: 34368338 PMCID: PMC8316968 DOI: 10.12998/wjcc.v9.i21.6155] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/29/2021] [Revised: 04/30/2021] [Accepted: 05/20/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND The inflammatory myofibroblastic tumor (IMT) is a rare, idiopathic, usually benign, mass-forming disease with myofibroblastic proliferation and a varying amount of inflammatory cells. Although it can affect various organs, the biliary tract is a rare localization of primary IMT, clinically, endoscopically and radiologically imitating cholangiocarcinoma. The treatment options are based only on clinical practice experience. CASE SUMMARY A 70-year-old woman was referred to our center due to progressive fatigue, weight loss, abdominal pain, night sweats, and elevated liver enzymes. Magnetic resonance cholangiopancreatography and endoscopic retrograde cholangiopancreatography (ERCP) revealed proximal common hepatic duct and hilar biliary strictures extending bilaterally to lobular bile ducts. Although initial clinical, endoscopic and radiological signs were typical for hilar cholangiocarcinoma, histological examination showed no signs of malignancy. In total, 8 biopsies using different approaches were performed (several biopsies from dominant stricture during ERCP and direct cholangioscopy; ultrasound-guided liver biopsy; diagnostic laparoscopy with liver and lymph node biopsies). Histological examination revealed signs of IMT, and the final diagnosis of biliary IMT was stated. Although IMT is usually a benign disease, in our case, it was complicated. All pharmacological treatment measures were ineffective. The patient still needs permanent stenting, suffers from recurrent infections and mechanical jaundice. Despite that, the patient already survived 24 mo. CONCLUSION IMT presenting with hilar biliary strictures is a unique diagnostic and clinical challenge as it is indistinguishable from cholangiocarcinoma, and there are no evidence-based treatment options. Our goal is to increase the understanding of this rare disease and its possible course.
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Affiliation(s)
- Sandra Strainiene
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | | | - Juozas Jarasunas
- Department of Radiology, Nuclear Medicine and Medical Physics, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Ilona Savlan
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Juozas Stanaitis
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Ieva Stundiene
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Tomas Strainys
- Clinic of Anesthesiology and Intensive Care Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
| | - Valentina Liakina
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
- Department of Chemistry and Bioengineering, Faculty of Fundamental Science, Vilnius Gediminas Technical University, Vilnius 10223, Lithuania
| | - Jonas Valantinas
- Clinic of Gastroenterology, Nephrourology and Surgery, Institute of Clinical Medicine, Vilnius University, Vilnius 01513, Lithuania
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Filips A, Maurer MH, Montani M, Beldi G, Lachenmayer A. Inflammatory myofibroblastic tumor of the liver: A case report and review of literature. World J Hepatol 2020; 12:170-183. [PMID: 32685109 PMCID: PMC7336290 DOI: 10.4254/wjh.v12.i4.170] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/29/2019] [Revised: 03/05/2020] [Accepted: 03/12/2020] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Inflammatory myofibroblastic tumors of the liver (IMTL) are extremely rare neoplasms and very little is known about their clinical presentation, pathogenesis, and biological behavior. Due to their absolute rarity, it is almost impossible to obtain a definite diagnosis without histological examination. Because of their intermediate biological behavior with the risk for local recurrence and metastases, surgical resection is recommend whenever IMTL is suspect.
CASE SUMMARY We herein present a case of an otherwise healthy 32-year-old woman who presented with intermittent fever, unclear anemia, malaise and right flank pain 4 mo postpartum. The liver mass in segment IVa/b was highly FDG avid in the positron emission tomography-computed tomography. Hepatic resection was performed achieving a negative resection margin and an immediate resolution of all clinical symptoms. Histological analysis diagnosed the rare finding of an inflammatory myofibroblastic tumor of the liver and revealed cytoplasmic anaplastic lymphoma kinase expression by immunohistochemistry. Twelve months follow-up magnetic resonance imaging showed no recurrence and no metastases in the fully recovered patient.
CONCLUSION IMTLs are extremely rare and difficult to diagnose. Due to their intermediate biological behavior, surgical resection should be perform whenever feasible and patients should be followed-up in order to detect recurrence and metastasis as early as possible.
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Affiliation(s)
- Alexandra Filips
- Department of Visceral Surgery and Medicine, Inselspital, Bern University Hospital, University of Bern, Bern 3010, Switzerland
| | - Martin H Maurer
- Department of Radiology, Inselspital, Bern University Hospital, University of Bern, Bern 3010, Switzerland
| | - Matteo Montani
- Institute of Pathology, Inselspital, University Hospital, University Bern, Bern 3010, Switzerland
| | - Guido Beldi
- Department of Visceral Surgery and Medicine, Inselspital, Bern University Hospital, University of Bern, Bern 3010, Switzerland
| | - Anja Lachenmayer
- Department of Visceral Surgery and Medicine, Inselspital, Bern University Hospital, University of Bern, Bern 3010, Switzerland
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11
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Watanabe J, Yamada S, Sasaguri Y, Guo X, Kurose N, Kitada K, Inagaki M, Iwagaki H. A Surgical Case of Inflammatory Myofibroblastic Tumor of the Liver: Potentially Characteristic Gross Features. Clin Med Insights Oncol 2019; 13:1179554919829498. [PMID: 30799968 PMCID: PMC6378515 DOI: 10.1177/1179554919829498] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/13/2018] [Accepted: 01/07/2019] [Indexed: 12/17/2022] Open
Abstract
We herein reported a very rare surgical case of inflammatory myofibroblastic tumor (IMT) of the liver, showing potentially unique and specific gross findings on its cut surface: our IMT demonstrated a relatively well-demarcated and partly infiltrative and likely extrahepatic (ie serosal) but not intrahepatic mass, appearing firm and hemorrhagic, and yellow-whitish in color. The patient, who was a woman in her early 70s with 2-year follow-up for lung cryptococcosis and traffic accident, incidentally presented with unenhanced and low-density, heterogeneous mass on abdominal dynamic CT in the peripheral right lobe of the liver. We could conclusively diagnose the current lesion as the hepatic IMT after thorough analyses including a wide panel of immunohistochemical antibodies. Despite that, all clinicians and pathologists should be aware that the potentially characteristic, extrahepatic gross feature of IMT of the liver might also be one of the powerful supplementary tools for reaching its correct diagnosis. One of our aims in the presented case report is to emphasize that the hepatic IMT should be considered clinicopathologically in the differential diagnosis of mass lesions on the liver.
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Affiliation(s)
- Jiro Watanabe
- Laboratory of Pathology, National Hospital Organization, Fukuyama Medical Center, Fukuyama, Japan
| | - Sohsuke Yamada
- Laboratory of Pathology, National Hospital Organization, Fukuyama Medical Center, Fukuyama, Japan
- Department of Pathology and Laboratory Medicine, Kanazawa Medical University, Ishikawa, Japan
| | - Yasuyuki Sasaguri
- Laboratory of Pathology, National Hospital Organization, Fukuyama Medical Center, Fukuyama, Japan
- Laboratory of Pathology, Fukuoka Tokushukai Hospital, Fukuoka, Japan
| | - Xin Guo
- Department of Pathology and Laboratory Medicine, Kanazawa Medical University, Ishikawa, Japan
| | - Nozomu Kurose
- Department of Pathology and Laboratory Medicine, Kanazawa Medical University, Ishikawa, Japan
| | - Kouji Kitada
- Department of Surgery, National Hospital Organization, Fukuyama Medical Center, Fukuyama, Japan
| | - Masaru Inagaki
- Department of Surgery, National Hospital Organization, Fukuyama Medical Center, Fukuyama, Japan
| | - Hiromi Iwagaki
- Department of Surgery, National Hospital Organization, Fukuyama Medical Center, Fukuyama, Japan
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12
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Yang X, Zhu J, Biskup E, Cai F, Li A. Inflammatory pseudotumors of the liver: experience of 114 cases. Tumour Biol 2015; 36:5143-8. [PMID: 25663462 DOI: 10.1007/s13277-015-3167-y] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/18/2014] [Accepted: 01/26/2015] [Indexed: 01/01/2023] Open
Abstract
Hepatic inflammatory pseudotumors (HIPT) are rare benign neoplasms with unknown etiology and a great potential for mimicry, challenging diagnostics, and treatment features. The aim of the study was to retrospectively analyze the imaging, pathological, and clinical features of HIPT in our large cohort of patients in order to increase the understanding and suggest a scoring system for treatment approaches. Retrospective study analyzed 114 HIPT cases recorded from July 2006 to July 2012, when surgery was performed. Data were compared with chi-square test. In our study population, the mean age was 53.14 ± 10.98 years, with 69 male and 45 female patients. Most presented symptoms were abdominal pain (59/144, 41.0 %), fever (48/114, 42.1 %), abdominal distension (35/144, 24.3 %), and weight loss (12/144, 8.3 %). Laboratory examinations were normal. Sixteen cases were HBsAg positive and 8 had liver cirrhosis. Most of the tumors were located in the right lobe (79/114, 69.3 %), 33 in the left lobe, and 2 in the caudal lobe. Three imaging modalities, such as ultrasonography (US), computed tomography (CT), and magnetic resonance imaging (MRI), were compared and showed significant differences in sensitivity and sensibility. HIPT diagnostics are challenging, and conservative treatment should be prioritized as soon as the diagnosis is made. CT and MRI seem to have comparable diagnostic sensitivity. We propose a guideline for consideration of operative approach.
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Affiliation(s)
- Xiaoyu Yang
- Division of Special Treatment II, Eastern Hepatobiliary Surgery Hospital, Second Military Medical University, Shanghai, 200438, China,
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