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Balaji K, Siddique SS, Khurana U, Goel G, Tandon A, Joshi D, Chaudhary N, Khurana AK, Goyal A. Cytohistological findings and diagnostic challenges in rare pediatric pulmonary mesenchymal malignancies: A report of two cases. Diagn Cytopathol 2024; 52:E88-E94. [PMID: 38235937 DOI: 10.1002/dc.25273] [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: 06/17/2023] [Revised: 01/02/2024] [Accepted: 01/03/2024] [Indexed: 01/19/2024]
Abstract
Pulmonary mesenchymal tumors are uncommon neoplasms and the data available on their clinical, cytohistomorphological, immunohistochemical, and molecular findings are limited, leading to difficulty in timely diagnosis and management. Case 1: A 12-year-old boy presented with a right endobronchial mass. Imprint smears from endobronchial biopsy revealed moderately pleomorphic spindle cell tumor arranged in fascicles and perivascular pattern with attached myxoid material showing occasional mitotic activity suggesting a cytological diagnosis of sarcoma. Biopsy also displayed similar morphology. Extensive immunohistochemistry (IHC) showed diffuse SMA, focal AE1/AE3, and diffuse ALK positivity along with a MIB/Ki67 index of 30%-40% leading to the diagnosis of inflammatory myofibroblastic tumor (IMT; Sarcoma grade). Case 2: A 8-year-old boy presented with a huge left-sided mass replacing the entire lung parenchyma and eroding adjoining ribs. Pleural fluid cytology revealed vague clusters of PAS-positive diastase-sensitive small atypical cells with associated inflammatory cells in the hemorrhagic background, suggesting a diagnosis of malignant small round blue cell tumor. Trucut biopsy from the mass showed spindled to round cells showing diffuse positivity with CD99 and BCL-2. Molecular studies with reverse transcription-PCR (RT-PCR) for SYT-SSX and EWS-FLI1 were negative for synovial sarcoma and Ewing's sarcoma, respectively. Given the clinical setting, PAS positivity, IHC, and molecular studies, the diagnosis of tumors of uncertain differentiation with the possibility of Ewing's sarcoma family of tumors (ESFT) with a translocation between EWS1 and other ETS-family members (ERG, FEV, ETV1, E1AF, etc.) was suggested.
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Affiliation(s)
| | | | - Ujjawal Khurana
- Department of Pathology and Lab Medicine, AIIMS Bhopal, Bhopal, India
| | - Garima Goel
- Department of Pathology and Lab Medicine, AIIMS Bhopal, Bhopal, India
| | - Ashwani Tandon
- Department of Pathology and Lab Medicine, AIIMS Bhopal, Bhopal, India
| | - Deepti Joshi
- Department of Pathology and Lab Medicine, AIIMS Bhopal, Bhopal, India
| | | | | | - Abhishek Goyal
- Department of Pulmonary Medicine, AIIMS Bhopal, Bhopal, India
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Ferrell LD, Kakar S, Terracciano LM, Wee A. Tumours and Tumour-Like Lesions. MACSWEEN'S PATHOLOGY OF THE LIVER 2024:842-946. [DOI: 10.1016/b978-0-7020-8228-3.00013-2] [Citation(s) in RCA: 1] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/03/2025]
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Logaswari M, Tony Lim KH, Tan D, Ong CAJ, Sittampalam K. Inflammatory myofibroblastic tumour of the rectum with a novel ALK fusion diagnosed on cytology: not just another submucosal GIST! Cytopathology 2022; 33:397-401. [PMID: 35118731 DOI: 10.1111/cyt.13108] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/12/2021] [Revised: 01/08/2022] [Accepted: 01/29/2022] [Indexed: 11/30/2022]
Abstract
Inflammatory myofibroblastic tumours (IMT) are spindle cell neoplasms most commonly seen in the lungs, with a wide variety of less common extrapulmonary sites including the mesentery, omentum, and intrabdominal sites. On cytological evaluation, these tumours can be difficult to diagnose, given the morphological mimics of other submucosal spindle cell neoplasms, which may be compounded by the relatively small amount of tissue and the uncommon nature of the diagnosis. Immunohistochemical staining and molecular studies for the Anaplastic lymphoma kinase (ALK) gene can prove useful for diagnosing this tumour. We present the cytological features of an IMT occurring in the rectum, the differential diagnoses, useful immunohistochemical staining patterns and the additional finding of a novel ALK-fusion in this entity.
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Affiliation(s)
- M Logaswari
- Department of Pathology, Singapore General Hospital, Singapore, Singapore
| | - Kiat Hon Tony Lim
- Department of Pathology, Singapore General Hospital, Singapore, Singapore
| | - Damien Tan
- Department of Gastroenterology and Hepatology, Singapore General Hospital, Singapore
| | - Chin-Ann Johnny Ong
- Department of Sarcoma, Peritoneal and Rare Tumours (SPRinT), Division of Surgery and Surgical Oncology, National Cancer Centre Singapore.,Department of Sarcoma, Peritoneal and Rare Tumours (SPRinT), Division of Surgery and Surgical Oncology, Singapore General Hospital.,Laboratory of Applied Human Genetics, Division of Medical Sciences, National Cancer Centre, Singapore.,SingHealth Duke-NUS Oncology Academic Clinical Program, Duke-NUS Medical School, Singapore.,Institute of Molecular and Cell Biology, A*STAR Research Entities, Singapore
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4
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Grosse A, Grosse C. Unexpected finding of inflammatory myofibroblastic tumour in two cytological specimens. Cytopathology 2020; 31:333-337. [PMID: 32416636 DOI: 10.1111/cyt.12851] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/02/2020] [Revised: 05/09/2020] [Accepted: 05/10/2020] [Indexed: 11/29/2022]
Affiliation(s)
- Alexandra Grosse
- Institute of Pathology and Molecular Pathology, University Hospital Zurich, Zurich, Switzerland
| | - Claudia Grosse
- Institute of Pathology and Molecular Pathology, University Hospital Zurich, Zurich, Switzerland.,Institute of Pathology, Kepler University Hospital, Linz, Austria
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Nigam N, Rajani SS, Rastogi A, Patil A, Agrawal N, Sureka B, Arora A, Bihari C. Inflammatory pseudotumors of the liver: Importance of a multimodal approach with the insistance of needle biopsy. J Lab Physicians 2020; 11:361-368. [PMID: 31929705 PMCID: PMC6943873 DOI: 10.4103/jlp.jlp_63_19] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022] Open
Abstract
CONTEXT: Inflammatory pseudotumor (IPT) of the liver is a rare, tumor-like lesion that is considered to be biologically benign but often mimics malignancy. AIMS: The aim of the study was construe clinicopathological features, imaging findings, differential diagnosis, management, and follow-up of IPT involving the liver. SETTINGS AND DESIGN: It is a retrospective study. SUBJECTS AND METHODS: Cases included were of IPT, diagnosed on histopathology, at our center from June 2009 to December 2016. Details studied were clinical presentation, imaging studies, laboratory investigations, pathological features, treatment, and follow-up of the cases and compared with reports in the literature. RESULTS: A total of cases of IPT included were 17. The age of the patients ranged from 21 to 62 years. Common presenting features were intermittent fever, upper abdominal pain, and weight loss. Radiological diagnosis varied from neoplastic (13) to infectious etiologies (4), with hepatocellular carcinoma being the most common differential (7/17). Laboratory investigations revealed leukocytosis, hyperbilirubinemia, raised transaminases, and raised serum alkaline phosphatase. Core biopsy of a tumor conceded increased fibrosis along with mixed inflammatory cell infiltrates. Eleven cases were managed conservatively and showed regression or complete recovery. Six patients underwent surgical resection. None of these had any recurrence in median follow-up of 22 months. CONCLUSIONS: IPT of the liver can masquerade as a fatality, either primary or metastatic. It will be well managed with conservative modalities and can avoid redundant hepatectomy, reserved for complicated cases. For this intent, accurate preoperative diagnosis is the requisite, and needle biopsy with or without fine-needle aspiration cytology plays as a significant rescuer in this field.
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Affiliation(s)
- Neha Nigam
- Department of Pathology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India
| | - Shikha Singh Rajani
- Department of Pathology, Institute of Liver and Biliary Sciences, New Delhi, India
| | - Archana Rastogi
- Department of Pathology, Institute of Liver and Biliary Sciences, New Delhi, India
| | - Anupama Patil
- Department of Clinical Haematology, Institute of Liver and Biliary Sciences, New Delhi, India
| | - Nikhil Agrawal
- Department of Hepato-Pancreato-Biliary Surgery, Institute of Liver and Biliary Sciences, New Delhi, India
| | - Binit Sureka
- Department of Radiology, Institute of Liver and Biliary Sciences, New Delhi, India
| | - Asit Arora
- Department of Hepato-Pancreato-Biliary Surgery, Institute of Liver and Biliary Sciences, New Delhi, India
| | - Chhagan Bihari
- Department of Pathology, Institute of Liver and Biliary Sciences, New Delhi, India
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Kaytez SK, Kavuzlu A, Oguz H. Laryngeal Inflammatory Myofibroblastic Tumor With Anemia and Thrombocytosis. EAR, NOSE & THROAT JOURNAL 2019; 100:224-226. [PMID: 31569971 DOI: 10.1177/0145561319874057] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/21/2023] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) of the larynx is a rare benign lesion that commonly occurs in the soft tissues. We present the first case with systemic manifestations of laryngeal IMT that was associated with hypochromic, microcytic-type anemia and thrombocytosis.
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Affiliation(s)
- Selda Kargın Kaytez
- Department of Otorhinolaryngology, Ankara Training and Research Hospital, Ankara, Turkey
| | - Ali Kavuzlu
- Department of Otorhinolaryngology, Ankara Training and Research Hospital, Ankara, Turkey
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Ghosh M, Islam N, Saha H, Mukhopadhyay M, Datta C, Saha K, Chatterjee U. Cytodiagnosis of inflammatory myofibroblastic tumor: A report of three cases in infants. Diagn Cytopathol 2018; 46:776-781. [PMID: 29673102 DOI: 10.1002/dc.23950] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/12/2018] [Revised: 03/09/2018] [Accepted: 04/09/2018] [Indexed: 01/07/2023]
Abstract
Inflammatory myofibroblastic tumor is a tumor of intermediate grade with a low rate of metastasis. The tumor often mimics malignancy. There is sparse literature available describing the cytological findings of this rare condition. It often presents in infancy and childhood as an intra-abdominal mass. Here, we describe the cytological findings of three cases of inflammatory myofibroblastic tumor in infants aged 10 months, 4 months, and 15 months, respectively. While the first two cases are fine needle aspiration cytology (FNAC) smears, the third case is that of an intraoperative imprint cytology. All the three smears showed low cellularity with small clusters of bland spindle cells, along with sprinkling of inflammatory cells, suggestive of an inflammatory myofibroblastic tumor. The diagnosis was later confirmed on histopathology and positive immunostaining for ALK.
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Affiliation(s)
| | | | - Hinglaj Saha
- Department of Pediatric Surgery, NRS Medical College, Kolkata, India
| | | | | | - Koushik Saha
- Department of Pediatric Surgery, NRS Medical College, Kolkata, India
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9
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Fernandez-Bussy S, Inaty H, Caviedes I, Labarca G, Vial MR, Majid A. Unusual diagnoses made by convex-probe endobronchial ultrasound-guided transbronchial needle aspiration. Pulmonology 2018; 24:300-306. [PMID: 29627400 DOI: 10.1016/j.pulmoe.2017.12.004] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/28/2017] [Revised: 09/06/2017] [Accepted: 12/14/2017] [Indexed: 12/25/2022] Open
Abstract
Endobronchial ultrasound-guided transbronchial needle biopsy (EBUS-TBNA) has proven to be an effective and minimally invasive tool to diagnose and stage lung cancer. However, its use for the diagnosis of rare mediastinal and lung pathologies has been rarely described. Hereby we describe a retrospective chart review of our EBUS-TBNA database for unusual diagnosis made between July 2012 and October 2016. Those conditions considered unusual for EBUS-TBNA diagnosis were identified and their medical records reviewed.
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Affiliation(s)
- S Fernandez-Bussy
- Interventional Pulmonology, Clínica Alemana de Santiago, Facultad de Medicina Clínica Alemana - Universidad del Desarrollo, Santiago, Chile.
| | - H Inaty
- Department of Pulmonary and Critical Care Medicine, Cleveland Clinic, Cleveland, OH, United States
| | - I Caviedes
- Interventional Pulmonology, Clínica Alemana de Santiago, Facultad de Medicina Clínica Alemana - Universidad del Desarrollo, Santiago, Chile
| | - G Labarca
- Facultad de Medicina, Universidad San Sebastian, Concepcion, Chile; Complejo Asistencial Dr. Victor Rios Ruiz, Los Angeles, Chile
| | - M R Vial
- Interventional Pulmonology, Clínica Alemana de Santiago, Facultad de Medicina Clínica Alemana - Universidad del Desarrollo, Santiago, Chile
| | - A Majid
- Division of Thoracic Surgery and Interventional Pulmonology, Beth Israel Deaconess Medical Center, Boston, MA, United States
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Alfieri R, Alaggio R, Ruol A, Castoro C, Cagol M, Michieletto S, Pomerri F, Acquaviva A, Ancona E. Hepatogastric Inflammatory Pseudotumor Presumably Deriving from Prior Amebic Infection. TUMORI JOURNAL 2018; 94:584-8. [DOI: 10.1177/030089160809400423] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
Abstract
Introduction Inflammatory pseudotumor is a rare entity with a clinical and radiographic presentation that is difficult to differentiate from malignancy. This is a case report of a large hepatogastric inflammatory pseudotumor that presumably developed from a prior amebic pseudocyst. Case Report A 14-year-old boy presented with increasing vomiting, epigastric pain, dysphagia, asthenia and weight loss. The clinical history included an amebic infection at the age of 2 months. Instrumental investigations revealed an 8 x 6 cm left subdiaphragmatic mass inseparable from the gastric fundus, which appeared to infiltrate the left hepatic lobe. Surgery disclosed a bulky mass adhering to the gastric fundus and left hepatic lobe that prompted total gastrectomy, resection of the second and third hepatic segments, and Roux-en-Y esophagojejunal loop anastomosis. Histology subsequently confirmed that this was a pseudocyst with a large calcified nucleus surrounded by myofibroblastic proliferation associated with a diffuse lymphoplasmacytic infiltrate affecting the gastric wall and hepatic parenchyma, hence the final diagnosis of inflammatory pseudotumor, presumably in response to a prior amebic pseudocyst. Conclusions Inflammatory pseudotumor is a rare entity that is seldom found in the stomach. The particular interest of the present case lies in the fact that it developed in the stomach and liver, presumably deriving from a previous amebic pseudocyst.
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Affiliation(s)
| | - Rita Alaggio
- Department of Medical-Diagnostic Sciences and Special Therapies
| | - Alberto Ruol
- Department of Surgery and Gastroenterology, Clinica Chirurgica 3
| | | | | | | | | | | | - Ermanno Ancona
- Department of Surgery and Gastroenterology, Clinica Chirurgica 3
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Chauhan RS, Sodhi KS, Nada R, Virk R, Mathew J. Pediatric inflammatory myofibroblastic tumor of the trachea: Imaging spectrum and review of literature. Lung India 2018; 35:516-519. [PMID: 30381563 PMCID: PMC6219126 DOI: 10.4103/lungindia.lungindia_405_17] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022] Open
Abstract
Inflammatory myofibroblastic tumor of the airway is a very uncommon benign primary neoplasm in pediatric age group with increased local recurrence rate and potential metastatic spread. We describe a case of a 6-year boy who was brought to the pediatric emergency with severe respiratory distress, dry cough, and stridor. Contrast-enhanced computed tomography and magnetic resonance imaging (MRI) of the neck showed a polypoidal mass lesion in the right anterolateral trachea causing significant airway narrowing. Bronchoscopic findings correlated with the imaging. The lesion was confirmed at surgery and was completely removed by surgical excision. Histopathology revealed an inflammatory myofibroblastic tumor. MRI findings of this entity in a child have not been reported before.
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Affiliation(s)
- Richa Singh Chauhan
- Department of Radiodiagnosis and Imaging, Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | - Kushaljit Singh Sodhi
- Department of Radiodiagnosis and Imaging, Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | - Ritambhara Nada
- Department of Histopathology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | - Ramandeep Virk
- Department of Otorhinolaryngology, Post Graduate Institute of Medical Education and Research, Chandigarh, India
| | - Joseph Mathew
- Department of Pediatrics, Post Graduate Institute of Medical Education and Research, Chandigarh, India
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Ferrell LD, Kakar S, Terracciano LM, Wee A. Tumours and Tumour-like Lesions of the Liver. MACSWEEN'S PATHOLOGY OF THE LIVER 2018:780-879. [DOI: 10.1016/b978-0-7020-6697-9.00013-3] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/04/2025]
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Abstract
Sclerosing angiomatoid nodular transformation (SANT) of the spleen is a morphologically distinctive lesion. Although the clinical course of SANT is benign, its reactive or neoplastic nature remains to be clarified. Furthermore, some investigators have suggested that SANT is related to IgG4 sclerosing lesion or inflammatory pseudotumor with stromal cells positive for Epstein-Barr virus (EBV). In this study, we assessed 22 cases of SANT derived from adult women. Clinical data and follow-up information were obtained by chart review. Immunohistochemical studies for IgG4, IgG, and CD21 stains and in situ hybridization to detect EBV-encoded small RNAs were performed. We also assessed genomic DNA extracted from paraffin-embedded tissue for human androgen-receptor α gene analysis using conventional and methylation-specific polymerase chain reaction methods. The median patient age was 41.5 years (range, 25 to 82 y). Most (77%) patients presented with a single mass that was detected incidentally (59%). The mean size of the lesions was 3.8 cm (range, 1.0 to 9.0 cm). Clinical symptoms correlated with multiple lesions (P=0.043) but not lesional size (P=0.637) or location in the spleen (hilum vs. periphery, P=0.696). None of the cases had evidence of IgG4-related disease or recurred after splenectomy. The mean number of IgG4 cells was 27.7 (range, 4 to 125), and the mean IgG4/IgG ratio was 16.4% (range, 1.6% to 55.7%) with only 2 cases being >40%. Cases with higher IgG4 cells did not correlate with inflammatory pseudotumor-like morphology. No lesions were positive for EBV-encoded small RNAs, and almost all cases with informative results (n=19) showed a polyclonal pattern. We conclude that SANT is a polyclonal, reactive lesion rather than a neoplasm.
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Sharma S, Gupta N, Rajwanshi A, Saikia UN. Inflammatory myofibroblastic tumour: a diagnostic challenge on fine needle aspiration cytology. Cytopathology 2016; 27:512-516. [DOI: 10.1111/cyt.12337] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/07/2016] [Indexed: 11/27/2022]
Affiliation(s)
- S. Sharma
- Department of Cytology and Gynaecological Pathology; Postgraduate Institute of Medical Education and Research; Chandigarh India
| | - N. Gupta
- Department of Cytology and Gynaecological Pathology; Postgraduate Institute of Medical Education and Research; Chandigarh India
| | - A. Rajwanshi
- Department of Cytology and Gynaecological Pathology; Postgraduate Institute of Medical Education and Research; Chandigarh India
| | - U. N. Saikia
- Department of Histopathology; Postgraduate Institute of Medical Education and Research; Chandigarh India
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16
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Lee JC, Wu JM, Liau JY, Huang HY, Lo CY, Jan IS, Hornick JL, Qian X. Cytopathologic features of epithelioid inflammatory myofibroblastic sarcoma with correlation of histopathology, immunohistochemistry, and molecular cytogenetic analysis. Cancer Cytopathol 2015; 123:495-504. [PMID: 26139079 DOI: 10.1002/cncy.21558] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/10/2015] [Revised: 04/18/2015] [Accepted: 04/21/2015] [Indexed: 12/17/2022]
Abstract
BACKGROUND Epithelioid inflammatory myofibroblastic sarcoma (E-IMS) is a recently established rare variant of inflammatory myofibroblastic tumor. It is characterized by a distinctive constellation of clinical, pathological, and molecular features, including a nearly exclusive intraabdominal location, strong male predilection, aggressive clinical course, predominance of epithelioid tumor cells, and Ran-binding protein 2 (RANBP2)-anaplastic lymphoma kinase (ALK) fusion in the majority of cases. To the authors' knowledge, the cytologic features of E-IMS have not been described to date. METHODS Cases of E-IMS that had corresponding cytology were searched. Six cytology samples (1 fine-needle aspiration sample, 2 imprint samples, and 3 effusion fluids) containing tumor cells were identified in 5 patients with E-IMS. RESULTS The cytomorphology included large monotonous epithelioid cells arranged in loose aggregates or singly, with admixed myxoid stroma, and an inflammatory background rich in neutrophils. The tumor cells had a large, round, eccentric nucleus with vesicular chromatin, prominent nucleoli, and moderate amounts of pale cytoplasm. Delicate thin-walled branching vessels traversing tumor aggregates was a prominent feature in a fine-needle aspiration sample. Immunohistochemically, ALK was positive in all 5 tumors, with a nuclear membranous staining pattern noted in 3 cases and a cytoplasmic pattern observed in the other 2 cases. ALK rearrangement was confirmed in all 5 tumors by molecular genetic studies. CONCLUSIONS The cytologic features of E-IMS recapitulate its histologic characteristics. E-IMS merits inclusion in the differential diagnosis of any intraabdominal, large epithelioid cell neoplasm. Confirmation of ALK rearrangement is advisable because patients may benefit from targeted therapies.
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Affiliation(s)
- Jen-Chieh Lee
- Department and Graduate Institute of Pathology, National Taiwan University Hospital, National Taiwan University College of Medicine, Taipei, Taiwan
| | - Jiann-Ming Wu
- Department of Surgery, Far Eastern Memorial Hospital, New Taipei City, Taiwan
| | - Jau-Yu Liau
- Department and Graduate Institute of Pathology, National Taiwan University Hospital, National Taiwan University College of Medicine, Taipei, Taiwan
| | - Hsuan-Ying Huang
- Department of Pathology, Kaohsiung Chang Gung Memorial Hospital, Chang Gung University College of Medicine, Kaohsiung, Taiwan
| | - Cheng-Yu Lo
- Department of Pathology, Far Eastern Memorial Hospital, New Taipei City, Taiwan
| | - I-Shiow Jan
- Department of Laboratory Medicine, National Taiwan University Hospital, National Taiwan University College of Medicine, Taipei, Taiwan
| | - Jason L Hornick
- Department of Pathology, Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - Xiaohua Qian
- Department of Pathology, Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
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Huang Y, Li LP, Wang J, Lun ZJ, Li W, Yang Z. Inflammatory pseudotumor of the colon causing intussusception: A case report and literature review. World J Gastroenterol 2015; 21:704-710. [PMID: 25593502 PMCID: PMC4292308 DOI: 10.3748/wjg.v21.i2.704] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/23/2014] [Revised: 06/18/2014] [Accepted: 07/30/2014] [Indexed: 02/06/2023] Open
Abstract
Inflammatory pseudotumor (IPT) is a rare space-occupying lesion of unknown etiology that can mimic malignancy on clinic-radiological and pathological examination. We describe a rare case of ileocecal intussusception from clinically suspected malignancy of the right colon where the patient underwent right hemicolectomy. Histopathology of the resected specimen confirmed IPT of the colon. This patient was observed to have abnormally elevated total leukocyte count and platelets before and after surgery. In an adult with intussusception associated with an abdominal mass, the possibility of IPT of the colon should be considered. Considering the abnormally high total leukocyte and platelet counts and colonic IPT, it is necessary to prevent postoperative adverse effects due to these changes. Although IPT of the colon is usually a benign process, controversy regarding its management still exists. We consider hemicolectomy as a safe treatment approach for colonic IPT and review the existing literature.
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Anbardar MH, Kumar PV, Hosseini SV. Fine needle aspiration cytology of inflammatory pseudotumour of the spleen. Cytopathology 2014; 26:202-4. [PMID: 24785340 DOI: 10.1111/cyt.12147] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
Affiliation(s)
- M H Anbardar
- Department of Pathology, Colorectal Research Centre, Shiraz Medical School, Shiraz University of Medical Sciences, Shiraz, Iran
| | - P V Kumar
- Department of Pathology, Colorectal Research Centre, Shiraz Medical School, Shiraz University of Medical Sciences, Shiraz, Iran
| | - S V Hosseini
- Department of Surgery, Colorectal Research Centre, Shiraz Medical School, Shiraz University of Medical Sciences, Shiraz, Iran
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Inflammatory pseudotumor of the thymus: A case report and review of the literature. Oncol Lett 2014; 7:1414-1418. [PMID: 24765147 PMCID: PMC3997730 DOI: 10.3892/ol.2014.1895] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/17/2013] [Accepted: 01/30/2014] [Indexed: 11/29/2022] Open
Abstract
Inflammatory pseudotumors are rare benign tumors consisting of cellular and stromal elements of a localized reactive process. While inflammatory pseudotumors are commonly detected in the lung and occasionally in other organs, only one case of inflammatory pseudotumor of the thymus has been reported in the literature to date. This report presents a 54-year-old male patient with inflammatory pseudotumor of the thymus accompanied by pulmonary inflammation. The patient presented with chest pain and moderate fever for 12 days. Enhanced computed tomography of the thorax revealed an anterior mediastinal solid and cystic mass, which constricted the left brachiocephalic vein accompanied by bilateral lung inflammation and marginal pleural effusion. The patient underwent a median sternotomy for the surgical removal of the mass. Histologically, the resected mass was composed of necrotic and fibrous tissues and inflammatory infiltrates, and the diagnosis was confirmed as an inflammatory pseudotumor of the thymus. The patient’s symptoms were resolved following surgery and the patient remained asymptomatic during the six-month follow-up period. In addition, we reviewed the previous literature and discussed the diagnosis and management of our patient. This report provides further insights into the pathogenesis and underlying mechanisms of inflammatory pseudotumors of the thymus to aid in the diagnosis and development of effective therapies.
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Inflammatory myofibroblastic tumor: a rarely seen submucosal lesion of the stomach. Case Rep Oncol Med 2013; 2013:328108. [PMID: 23573435 PMCID: PMC3615588 DOI: 10.1155/2013/328108] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/30/2013] [Accepted: 02/24/2013] [Indexed: 11/18/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal benign tumor which is generally seen in children and in young adults. It is especially located in the lungs. In histopathological examination, neoplastic fusiform cells originating from a subtype of accessory immune system cells which are called fibroblastic reticulum cells are seen (Kouichi and Youichirou, 2008). Although IMT is histopathologically benign, imaging methods show its tendency for local recurrence and invasion. In most of the cases, it may not be possible to make a distinction whether it is malign or benign. Complete surgical resection is the most important treatment method. In this study, we have discussed the findings of our case having a gastric submucosal located IMT in light of the current literatures.
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Bal MM, Deodhar K, Shrikhande S, Shukla P, Arya S, Ramadwar M. Solid pseudopapillary tumor of the pancreas: ‘Experiences’ and ‘Lessons’ at a tertiary-care oncology center. Diagn Cytopathol 2012; 41:599-606. [DOI: 10.1002/dc.22908] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/02/2011] [Accepted: 06/29/2012] [Indexed: 11/10/2022]
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Goodman ZD, Terracciano LM, Wee A. Tumours and tumour-like lesions of the liver. MACSWEEN'S PATHOLOGY OF THE LIVER 2012:761-851. [DOI: 10.1016/b978-0-7020-3398-8.00014-3] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/04/2025]
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23
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Mundi I, Singhal N, Punia RPS, Dalal U, Mohan H. Inflammatory pseudotumor of the spleen: a rare case diagnosed on FNAC. Diagn Cytopathol 2011; 40:1104-6. [PMID: 21563321 DOI: 10.1002/dc.21719] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/18/2010] [Accepted: 03/15/2011] [Indexed: 01/03/2023]
Abstract
Inflammatory pseudotumor (IPT) is a rare space-occupying lesion of unknown etiology that can mimic malignancy on clinic-radiological and pathological examination. We present a case of IPT of the spleen which was clinically suspected to be malignant. This case was initially suggested accurately on fine needle aspiration (FNA) of the mass and subsequently confirmed on histopathology of the resected specimen.
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Affiliation(s)
- Irneet Mundi
- Department of Pathology, Government Medical College and Hospital, Chandigarh, India
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Borak S, Siegal GP, Reddy V, Jhala N, Jhala D. Metastatic inflammatory myofibroblastic tumor identified by EUS-FNA in mediastinal lymph nodes with ancillary FISH studies for ALK rearrangement. Diagn Cytopathol 2011; 40 Suppl 2:E118-25. [PMID: 21472870 DOI: 10.1002/dc.21663] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/22/2010] [Accepted: 01/12/2011] [Indexed: 12/28/2022]
Affiliation(s)
- Samuel Borak
- Department of Pathology, University of Alabama at Birmingham, AL, USA
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25
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Stoll LM, Li QK. Cytology of fine-needle aspiration of inflammatory myofibroblastic tumor. Diagn Cytopathol 2010; 39:663-72. [PMID: 20730898 DOI: 10.1002/dc.21444] [Citation(s) in RCA: 24] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/08/2010] [Accepted: 04/17/2010] [Indexed: 12/25/2022]
Affiliation(s)
- Lisa Marie Stoll
- Division of Cytopathology, Department of Pathology, The Johns Hopkins Hospitals, Baltimore, Maryland, USA
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Al-Jabri T, Sanjay P, Shaikh I, Woodward A. Inflammatory myofibroblastic pseudotumour of the liver in association with gall stones - a rare case report and brief review. Diagn Pathol 2010. [PMID: 20718986 DOI: 10.1186/1746-1596-5-53al-jabri,] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023] Open
Abstract
Inflammatory myofibroblastic pseudotumours of the liver are rare tumour-like lesions that can mimic malignant liver neoplasms. The symptoms and radiological findings of this rare tumour can pose diagnostic difficulties. We describe a 69-year-old gentleman who was admitted to our department with symptoms suggestive of acute cholecystitis. Ultrasonography and computed tomography of the liver raised the possibility of metastatic liver disease. A core biopsy of the liver was performed to confirm the diagnosis of liver metastasis. Unexpectedly it showed no evidence of malignancy but instead revealed an inflammatory myofibroblastic pseudotumour of the liver. This case report highlights the diagnostic dilemma that arose due to the similarity of appearances between the two pathological entities on imaging and this stresses the need for accurate histological diagnosis so as to avoid unnecessary surgical intervention. To the best of our knowledge, only a minority of cases are reported in the literature associating a hepatic inflammatory myofibroblastic pseudotumour with gall stones.
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Affiliation(s)
- Talal Al-Jabri
- Department of Surgery, East and North Hertforshire NHS Trust, Hertfordshire, AL7 4HQ, UK.
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Al-Jabri T, Sanjay P, Shaikh I, Woodward A. Inflammatory myofibroblastic pseudotumour of the liver in association with gall stones - a rare case report and brief review. Diagn Pathol 2010; 5:53. [PMID: 20718986 PMCID: PMC2933681 DOI: 10.1186/1746-1596-5-53] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/09/2010] [Accepted: 08/18/2010] [Indexed: 01/17/2023] Open
Abstract
Inflammatory myofibroblastic pseudotumours of the liver are rare tumour-like lesions that can mimic malignant liver neoplasms. The symptoms and radiological findings of this rare tumour can pose diagnostic difficulties. We describe a 69-year-old gentleman who was admitted to our department with symptoms suggestive of acute cholecystitis. Ultrasonography and computed tomography of the liver raised the possibility of metastatic liver disease. A core biopsy of the liver was performed to confirm the diagnosis of liver metastasis. Unexpectedly it showed no evidence of malignancy but instead revealed an inflammatory myofibroblastic pseudotumour of the liver. This case report highlights the diagnostic dilemma that arose due to the similarity of appearances between the two pathological entities on imaging and this stresses the need for accurate histological diagnosis so as to avoid unnecessary surgical intervention. To the best of our knowledge, only a minority of cases are reported in the literature associating a hepatic inflammatory myofibroblastic pseudotumour with gall stones.
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Affiliation(s)
- Talal Al-Jabri
- Department of Surgery, East and North Hertforshire NHS Trust, Hertfordshire, AL7 4HQ, UK.
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Pitman MB. Liver. Diagn Cytopathol 2010. [DOI: 10.1016/b978-0-7020-3154-0.00008-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/24/2022]
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29
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Sivanandan S, Lodha R, Agarwala S, Sharma M, Kabra SK. Inflammatory myofibroblastic tumor of the trachea. Pediatr Pulmonol 2007; 42:847-50. [PMID: 17623857 DOI: 10.1002/ppul.20651] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/08/2022]
Abstract
Inflammatory myofibroblastic tumor of the trachea is a rare benign tumor in children. We describe a 9-year-old girl who presented with recurrent episodes of wheeze and severe respiratory distress requiring mechanical ventilation. She had recurrent collapses of the right lung and a chest CT and bronchoscopy confirmed the presence of an obstructing mass lesion at the carina. The lesion, 1.5 cm x 1.5 cm in size, was debulked by rigid bronchoscopy and histopathology revealed features of inflammatory myofibroblastic tumor. A repeat bronchoscopy at 1 month of follow up did not reveal any residual lesion.
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Affiliation(s)
- Sindhu Sivanandan
- Department of Pediatrics, All India Institute of Medical Sciences, Ansari Nagar, New Delhi 110029, India
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Hoosein MM, Tapuria N, Standish RA, Koti RS, Webster GJM, Millar AD, Davidson BR. Inflammatory pseudotumour of the liver: the residuum of a biliary cystadenoma? Eur J Gastroenterol Hepatol 2007; 19:333-6. [PMID: 17353699 DOI: 10.1097/meg.0b013e328010b128] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/10/2022]
Abstract
Inflammatory pseudotumour is a rare form of a liver mass. We report the case of a 28-year-old man presenting with obstructive jaundice, in whom an inflammatory pseudotumour arose with the resolution of a mucus secreting cystic liver lesion. The initial features suggested an intrahepatic cystadenoma or cystadenocarcinoma, which on its involution left a solid mass. Histopathology showed an inflammatory pseudotumour with no evidence of malignancy. A similar case has been reported recently, with the development of an inflammatory pseudotumour following collapse of a liver cyst seen on imaging. These two cases may shed some light on the origins of these rare liver lesions.
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Affiliation(s)
- Moinuddin M Hoosein
- University Department of Surgery, Royal Free and University College Medical School, Royal Free Hospital, London, UK
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Nakamura H, Kawasaki N, Taguchi M, Kitamura H. Pulmonary inflammatory myofibroblastic tumor resected by video-assisted thoracoscopic surgery: Report of a case. Surg Today 2007; 37:137-40. [PMID: 17243033 DOI: 10.1007/s00595-006-3344-0] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/27/2006] [Accepted: 07/09/2006] [Indexed: 12/17/2022]
Abstract
Pulmonary inflammatory myofibroblastic tumor (IMT) is rare. A 38-year-old woman visited our hospital 2 days after experiencing transient anterior chest pain. Chest radiography showed a nodule suggestive of adenocarcinoma in the middle lobe; however, transbronchial lung biopsy and computed tomography (CT)-guided percutaneous needle cytology showed signs of inflammation. To obtain a definitive diagnosis we performed a wedge resection using video-assisted thoracoscopic surgery and removed the nodule completely. The pathologic diagnosis made during surgery was IMT. The longest dimension of the nodule was 28 mm. Immunohistochemical examination showed proliferating spindle cells, which were positive for vimentin and smooth muscle actin, but negative for desmin, CD34, cytokeratin, epithelial membrane antigen, S100 protein, and neuron specific enolase. These findings were consistent with the staining pattern of IMT previously reported. Careful follow-up is necessary to detect any sign of local recurrence and distant metastases.
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Affiliation(s)
- Haruhiko Nakamura
- Departments of Chest Surgery, Atami Hospital, International University of Health and Welfare, 13-1 Higashikaigan-cho, Atami, Shizuoka, 413-0012, Japan
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Solomon GJ, Kinkhabwala MM, Akhtar M. Inflammatory myofibroblastic tumor of the liver. Arch Pathol Lab Med 2006; 130:1548-51. [PMID: 17090200 DOI: 10.5858/2006-130-1548-imtotl] [Citation(s) in RCA: 16] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 03/16/2006] [Indexed: 11/06/2022]
Abstract
We report a case of hepatic inflammatory myofibroblastic tumor in a 26-year-old African American man who presented with right upper quadrant pain, weight loss, and fatigue during the previous year. Hepatomegaly was found on physical examination. Laboratory findings were significant for mild normocytic, normochromic anemia and elevated erythrocyte sedimentation rate. Imaging studies showed 2 contiguous masses suspicious for malignancy. A left partial hepatectomy was performed; the preoperative differential diagnosis was for angiosarcoma and hepatocellular carcinoma. The resected liver specimen showed 2 contiguous, firm, tan-white nodules that microscopically represented a proliferation of spindled myofibroblast cells set in an inflammatory and collagenized background. The spindle cells were strongly reactive for smooth muscle actin but negative for ALK-1. The morphologic and immunophenotypic findings, coupled with the clinical presentation, were consistent with an inflammatory myofibroblastic tumor of the liver.
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Affiliation(s)
- Garron J Solomon
- New York-Presbyterian Hospital, Weill Cornell Medical College, New York, NY 10021, USA.
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Neild GH, Rodriguez-Justo M, Wall C, Connolly JO. Hyper-IgG4 disease: report and characterisation of a new disease. BMC Med 2006; 4:23. [PMID: 17026742 PMCID: PMC1618394 DOI: 10.1186/1741-7015-4-23] [Citation(s) in RCA: 190] [Impact Index Per Article: 10.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/17/2006] [Accepted: 10/06/2006] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND We highlight a chronic inflammatory disease we call 'hyper-IgG4 disease', which has many synonyms depending on the organ involved, the country of origin and the year of the report. It is characterized histologically by a lymphoplasmacytic inflammation with IgG4-positive cells and exuberant fibrosis, which leaves dense fibrosis on resolution. A typical example is idiopathic retroperitoneal fibrosis, but the initial report in 2001 was of sclerosing pancreatitis. METHODS We report an index case with fever and severe systemic disease. We have also reviewed the histology of 11 further patients with idiopathic retroperitoneal fibrosis for evidence of IgG4-expressing plasma cells, and examined a wide range of other inflammatory conditions and fibrotic diseases as organ-specific controls. We have reviewed the published literature for disease associations with idiopathic, systemic fibrosing conditions and the synonyms: pseudotumour, myofibroblastic tumour, plasma cell granuloma, systemic fibrosis, xanthofibrogranulomatosis, and multifocal fibrosclerosis. RESULTS Histology from all 12 patients showed, to varying degrees, fibrosis, intense inflammatory cell infiltration with lymphocytes, plasma cells, scattered neutrophils, and sometimes eosinophilic aggregates, with venulitis and obliterative arteritis. The majority of lymphocytes were T cells that expressed CD8 and CD4, with scattered B-cell-rich small lymphoid follicles. In all cases, there was a significant increase in IgG4-positive plasma cells compared with controls. In two cases, biopsies before and after steroid treatment were available, and only scattered plasma cells were seen after treatment, none of them expressing IgG4. Review of the literature shows that although pathology commonly appears confined to one organ, patients can have systemic symptoms and fever. In the active period, there is an acute phase response with a high serum concentration of IgG, and during this phase, there is a rapid clinical response to glucocorticoid steroid treatment. CONCLUSION We believe that hyper-IgG4 disease is an important condition to recognise, as the diagnosis can be readily verified and the outcome with treatment is very good.
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Affiliation(s)
- Guy H Neild
- UCL Centre for Nephrology, Royal Free Hospital, London NW3 2QG, UK
- Institute of Urology and Nephrology, Middlesex Hospital, London W1T 3AA, UK
| | - Manuel Rodriguez-Justo
- Department of Histopathology, Royal Free and University College Medical School, University College Hospital, Rockefeller Building, London WC1E 6JJ, UK
| | - Catherine Wall
- UCL Centre for Nephrology, Royal Free Hospital, London NW3 2QG, UK
| | - John O Connolly
- UCL Centre for Nephrology, Royal Free Hospital, London NW3 2QG, UK
- Institute of Urology and Nephrology, Middlesex Hospital, London W1T 3AA, UK
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Sen G, Tiwari M, Gupta S. Large plasma cell tumour of the lung in a young adult—A case report. Indian J Thorac Cardiovasc Surg 2006. [DOI: 10.1007/s12055-006-0025-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/22/2022] Open
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