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Dwivedi S, Rakesh CR, Anand S, Dogra N, Singh BP. Intra-abdominal Inflammatory Myofibroblastic Tumour (IMFT)-Uncommon Entity. Indian J Surg Oncol 2024; 15:344-348. [PMID: 38817997 PMCID: PMC11133245 DOI: 10.1007/s13193-023-01869-8] [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: 11/23/2023] [Accepted: 12/18/2023] [Indexed: 06/01/2024] Open
Abstract
IMFT (inflammatory myofibroblastic tumour) is an uncommon tumour predominantly affecting the lungs and mediastinum. Most of the published literature supports that it affects children and young individuals. IMFT involving the gastrointestinal tract is rare. We report a case of multifocal IMFT affecting the GI tract which was managed with gross total excision followed by chemotherapy. Surgical resection remains the treatment of choice. The role of chemotherapy and radiation therapy remains limited. The aetiology of these tumours remains unclear and is mostly ALK-positive that could be targeted. Local recurrences are common and hence require close follow-up. The risk of recurrences and metastasis is increased in cases with TP53 positivity, aneuploidy and recurrent lesions.
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Affiliation(s)
| | | | - S. Anand
- Command Hospital Air Force, Bangalore, India
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2
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A G H, Kumar S, Singla S, Kurian N. Aggressive Inflammatory Myofibroblastic Tumor of Distal Pancreas: A Diagnostic and Surgical Challenge. Cureus 2022; 14:e22820. [PMID: 35399449 PMCID: PMC8980218 DOI: 10.7759/cureus.22820] [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: 03/03/2022] [Indexed: 11/05/2022] Open
Abstract
An inflammatory myofibroblastic tumor (IMT) is a rare soft tissue neoplasm of unknown etiology. It is a slow-growing tumor of borderline malignant potential. Distant metastases and recurrence after complete excision are rare. Establishing a preoperative diagnosis is difficult because of its nonspecific clinic-radiological features. Although the majority of cases have been reported in the lungs, it can affect any part of the body. The pancreatic inflammatory myofibroblastic tumor is very rare and only 26 cases have been reported in the medical literature. These tumors mostly arise from the head of the pancreas, whereas occurrence in the body or tail region is rather unusual. Here, we report a case of a 55-year-old male patient with a locally advanced inflammatory myofibroblastic tumor arising from the pancreatic tail. Complete excision of tumor required multi-visceral resection (distal pancreaticosplenectomy with jejunal and colonic segmental resection). The diagnosis of inflammatory myofibroblast tumor was made on the basis of histopathology and immunohistochemistry.
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Chen ZT, Lin YX, Li MX, Zhang T, Wan DL, Lin SZ. Inflammatory myofibroblastic tumor of the pancreatic neck: A case report and review of literature. World J Clin Cases 2021; 9:6418-6427. [PMID: 34435007 PMCID: PMC8362560 DOI: 10.12998/wjcc.v9.i22.6418] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/18/2021] [Revised: 03/23/2021] [Accepted: 06/01/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Pancreatic inflammatory myofibroblastic tumor (IMT) is a relatively rare disease that is often confused with pancreatic cancer or pancreatic neuroendocrine tumors. The histological features of IMTs show that tissue from this type of tumor contains an intermingling of fibroblast and myofibroblast proliferation, accompanied by a varying degree of inflammatory cell infiltration.
CASE SUMMARY The management of an IMT occurring at the neck of the pancreas is presented in this paper. A 66-year-old female patient was diagnosed with a pancreatic neck mass after a series of tests. The patient underwent enucleation of the pancreatic neck tumor after a pathological diagnosis of IMT. Previous research on the clinical features, pathological diagnosis and treatment of pancreatic IMTs was reviewed. Compared with previous reports, this is a unique case of enucleation of a pancreatic IMT.
CONCLUSION The enucleation of pancreatic IMTs may be a safe and efficient surgical method for managing such tumors with a better prognosis. Further cases are required to explore surgical measures for pancreatic IMTs.
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Affiliation(s)
- Zhi-Tao Chen
- Department of Hepatobiliary and Pancreatic Surgery, Shulan (Hangzhou) Hospital Affiliated to Zhejiang Shuren University Shulan International Medical College, Hangzhou 310000, Zhejiang Province, China
- School of Medicine, Zhejiang University, Hangzhou 310000, Zhejiang Province, China
| | - Yao-Xiang Lin
- School of Medicine, Hangzhou Normal University, Hangzhou 310003, Zhejiang Province, China
| | - Meng-Xia Li
- School of Medicine, Zhejiang University, Hangzhou 310000, Zhejiang Province, China
- Department of Hepatobiliary and Pancreatic Surgery, First Affiliated Hospital, School of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
| | - Ting Zhang
- Department of Pathology, Shulan (Hangzhou) Hospital Affiliated to Zhejiang Shuren University Shulan International Medical College, Hangzhou 310000, Zhejiang Province, China
| | - Da-Long Wan
- Department of Hepatobiliary and Pancreatic Surgery, The First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
| | - Sheng-Zhang Lin
- Department of Hepatobiliary Surgery, Shulan (Hangzhou) Hospital Affiliated to Zhejiang Shuren University Shulan International Medical College, Hangzhou 310000, Zhejiang Province, China
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A Pancreatic Inflammatory Myofibroblastic Tumor with Spontaneous Remission: A Case Report with a Literature Review. Diagnostics (Basel) 2019; 9:diagnostics9040150. [PMID: 31627359 PMCID: PMC6963339 DOI: 10.3390/diagnostics9040150] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/25/2019] [Revised: 10/13/2019] [Accepted: 10/15/2019] [Indexed: 12/17/2022] Open
Abstract
The inflammatory myofibroblastic tumor (IMT) is a rare tumor that can develop in any systemic organ. Its features are generally benign, but it often resembles malignancies and is treated surgically. Our patient was an 82-year-old female complaining of abdominal discomfort. Computed tomography demonstrated a 5 cm, ill-enhanced mass at the pancreas head. Upper gastrointestinal endoscopy revealed a duodenal submucosal tumor with apical erosion. Endoscopic ultrasonography (EUS) demonstrated a heterogeneous, low-echoic pancreas mass without clear margins. Fine-needle aspiration biopsy (FNAB) demonstrated spindle myofibroblastic tissues with lymphoplasmacyte and eosinophil infiltration, confirming an IMT diagnosis. Surprisingly, the tumor spontaneously regressed in one month without medication. Histological diagnosis using EUS-FNAB is essential for the rare pancreatic solid tumor like IMT.
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Qian J, Zhu K, Ye J. Ultrasonic Manifestations of Mesenteric Inflammatory Myofibroblastic Tumors in Children. Front Pediatr 2019; 7:39. [PMID: 30891434 PMCID: PMC6411639 DOI: 10.3389/fped.2019.00039] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/20/2018] [Accepted: 01/30/2019] [Indexed: 11/30/2022] Open
Abstract
Objective: To explore the ultrasonic manifestations of mesenteric inflammatory myofibroblastic tumors (IMTs) in children. Methods: Seven patients with mesenteric IMTs were retrospectively analyzed. The ultrasonic manifestations, such as the locations, sizes, morphology, borders, internal echo, blood flow, and metastasis, of the tumors were detected. Results: In all the seven pediatric patients, a solitary lesion was found for the mesenteric IMTs, including five cases in the ileocecal mesentery and two cases in the mesentery of ascending colon. All the single tumors were revealed as irregular hypoechoic masses with uneven internal echoes and enhanced echoes in the surrounding intestine and omentum. Internal blood flow signals were enriched in the tumors. The borders were clear in five cases and unclear in two cases. In addition, two cases had peritoneal effusion and one case had calcified plaques. In the follow-up studies, one of the seven IMT patients had malignant transformation, and one case was transferred to the pelvic cavity. Conclusion: Ultrasonic examination can clearly demonstrate the locations, sizes, morphology, borders, internal echo, blood flow as well as metastasis of the pediatric IMT of the mesentery, having an important clinical application value as an adjunct to computed tomography (CT).
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Affiliation(s)
- Jingjing Qian
- Department of Ultrasound, Children's Hospital, Zhejiang University School of Medicine, Hangzhou, China
| | - Kun Zhu
- Department of Pathology, The Children's Hospital, Zhejiang University School of Medicine, Hangzhou, China
| | - Jingjing Ye
- Department of Ultrasound, Children's Hospital, Zhejiang University School of Medicine, Hangzhou, China
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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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Tregnago AC, Morbeck DL, D’Almeida Costa F, Campos AHJFM, Soares FA, Vassallo J. Inflammatory pseudotumor-like follicular dendritic cell tumor: an underdiagnosed neoplasia. ACTA ACUST UNITED AC 2017. [DOI: 10.1186/s41241-017-0051-7] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
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Guffey Johnson J, Margo CE. Intraocular inflammatory mass associated with lens-induced uveitis. Surv Ophthalmol 2017; 62:541-545. [PMID: 28069494 DOI: 10.1016/j.survophthal.2016.12.013] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/16/2016] [Revised: 12/29/2016] [Accepted: 12/30/2016] [Indexed: 01/30/2023]
Abstract
Intraocular inflammatory tumefactions large enough to simulate neoplasms are uncommon. We report a patient with a large intraocular inflammatory mass composed of cells with features of histiocytes and myofibroblasts that was associated with lens-induced uveitis. The spindle cell mass appears to have arisen as an exaggerated response to exposed lens fibers. Although information from immunohistochemistry and cytogenetics has advanced the classification of inflammatory tumefactions, this case highlights the challenges in establishing the nature of these lesions.
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Affiliation(s)
- Jean Guffey Johnson
- Department of Pathology, James A. Haley Veterans Affairs Hospital, Tampa, Florida, USA
| | - Curtis E Margo
- Department of Pathology and Cell Biology, Morsani College of Medicine, University of South Florida, Tampa, Florida, USA; Department of Ophthalmology, Morsani College of Medicine, University of South Florida, Tampa, Florida, USA.
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Abstract
Over the past three decades, Immunohistochemistry has materially changed the practice of diagnostic surgical pathology. Foundational observations in this field were critical to a reasoned assessment of both the risks and opportunities that immunohistochemistry afforded the surgical pathologist, and our current practice draws heavily on those early assessments. As we collectively look to and acknowledge those who recognized the value of this technique and who helped guide its development as a companion to (not a replacement for) histomorphologic evaluation, we are drawn to those whose mastery of detail and ability to draw common patterns from seemingly unrelated phenomena helped define the diagnostic power of immunohistochemistry. The focus of this review is on one individual, Dr. Juan Rosai, whose contributions transcend the simple linkage of molecular observations to morphology, recognizing novel patterns in both form and color (the latter often the lovely shades of diaminobenzidine), seemingly viewing our diagnostic world at times through an entirely different lens. By looking at Dr. Rosai's early work in this field, reviewing a selection of his seminal observations, particularly in the Immunohistochemistry of thyroid and thymic neoplasia, revisiting how his special insight is often guided by the work of the early masters of morphology, and how his mentorship of others has helped shaped academic surgical pathology practice, perhaps we can get a glimpse through that lens.
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Affiliation(s)
- Paul E Swanson
- Cumming School of Medicine, Calgary Laboratory Services, Calgary, Alberta.
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10
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Xu LF, Zhou J, Liang CZ. Inflammatory Myofibroblastic Tumor of Genitourinary Tract Beyond Collecting System: A Rare Case Report With Literature Review. Medicine (Baltimore) 2015; 94:e1706. [PMID: 26496281 PMCID: PMC4620822 DOI: 10.1097/md.0000000000001706] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/25/2023] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) rarely arises in genitourinary tract especially beyond collecting system, which determines the unspecific clinic symptoms and sometimes can mimic malignancy. Therefore, IMT's diagnosis may usually be a pitfall. This case report characterizes a 35-year-old woman with a history of lower quadrant lasting pain followed by fever. Furthermore, radiologic examinations revealed that there were 2 lesions located in left adrenal area and left renalis. Owing to the anatomic complexity, the surgical resection was not complete. The pathologic diagnosis of the lesions was IMT. Adjuvant nonsteroids anti-inflammatory drugs were administrated after the operation. The symptoms were controlled finally and no further growing lesion was observed during a 1-year follow-up.Inflammatory myofibroblastic tumor is rare in genitourinary tract beyond the collecting system. Diagnosis should be based on histopathology. Presently, the authors report this rare case with the aim to share the experience regarding differential diagnosis and therapy.
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Affiliation(s)
- Ling-Fan Xu
- From the Department of Urology, The First Affiliated Hospital of Anhui Medical University, Hefei, China (LFX, JZ, C-ZL)
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11
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Cheng KJ, Wang SQ, Zhou SH. A case report of an inflammatory myofibroblastic tumor of the neck: A focus on the computed tomography and magnetic resonance imaging findings. Oncol Lett 2015; 10:518-522. [PMID: 26171061 DOI: 10.3892/ol.2015.3168] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/26/2014] [Accepted: 04/09/2015] [Indexed: 11/06/2022] Open
Abstract
Inflammatory myofibroblastic tumors (IMTs) of the neck are rare, with only a few patients reported in the literature. The present study discusses the clinical manifestations, radiographic characteristics and management of these tumors, with a focus on imaging modalities. A case of IMT of the neck is presented and the associated literature is reviewed. In total, seven patients in seven English-language studies, including the present case, and one patient in one Chinese-language study were found. On CT scans, all tumors appeared as soft-tissue densities. Upon magnetic resonance imaging (MRI), all tumors displayed a heterogeneous hypointense-isointense signal on T1-weighted sequences and an isointense-hyperintense signal on T2-weighted sequences. All tumors showed enhancement on enhanced CT and MR images. The imaging features of the neck IMTs can be summarized as follows: i) When enhanced, the tumor displays enhancement on CT and MR images; ii) MRI is superior to CT scans in the differential diagnosis of this disease; iii) in general, the lesion displays a hypointense-isointense signal on T1-weighted sequences and an isointense-hyperintense signal on T2-weighted sequences; iv) due to the fibrous tissue in the tumor, delayed enhancement may be observed on gadolinium-enhanced MR images; and v) due to its benign or intermediate features, the tumor is usually a well-defined mass.
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Affiliation(s)
- Ke-Jia Cheng
- Department of Otolaryngology, The First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, Zhejiang 310003, P.R. China
| | - Shen-Qing Wang
- Department of Otolaryngology, The First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, Zhejiang 310003, P.R. China
| | - Shui-Hong Zhou
- Department of Otolaryngology, The First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, Zhejiang 310003, P.R. China
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12
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Mid common bile duct inflammatory pseudotumor mimicking cholangiocarcinoma. A case report and literature review. Int J Surg Case Rep 2013; 5:12-5. [PMID: 24394855 PMCID: PMC3907201 DOI: 10.1016/j.ijscr.2013.10.017] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/20/2013] [Revised: 10/29/2013] [Accepted: 10/29/2013] [Indexed: 01/03/2023] Open
Abstract
INTRODUCTION Biliary inflammatory pseudotumors (IPTs) represent an exceptional benign cause of obstructive jaundice. These lesions are often mistaken for cholangiocarcinomas and are treated with major resections, because their final diagnosis can be achieved only after formal pathological examination of the resected specimen. Consequently, biliary IPTs are usually managed with unnecessary major resections. PRESENTATION OF CASE A 71-year-old female patient underwent an extra-hepatic bile duct resection en-bloc with the gallbladder and regional lymph nodes for an obstructing intraluminal growing tumor of the mid common bile duct (CBD). Limited resection was decided intraoperatively because of negative for malignancy fast frozen sections analysis in addition to the benign macroscopic features of the lesion. Histologically the tumor proved an IPT, arising from the bile duct epithelium, composed of inflammatory cells and reactive mesenchymal tissues. DISCUSSION The present case underlines the value of intraoperative reassessment of patients undergoing surgical resection for histopathologically undiagnosed biliary occupying lesions, in order to optimize their surgical management. CONCLUSION The probability of benign lesions mimicking cholangiocarcinoma should always be considered to avoid unnecessary major surgical resections, especially in fragile and/or elderly patients.
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13
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Radiological and histopathological features of hepatic inflammatory myofibroblastic tumour: Analysis of 10 cases. Clin Radiol 2013; 68:1114-20. [DOI: 10.1016/j.crad.2013.05.097] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/28/2013] [Revised: 05/19/2013] [Accepted: 05/29/2013] [Indexed: 01/17/2023]
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Inflammatory myofibroblastic bladder tumor in a patient with wolf-hirschhorn syndrome. Case Rep Urol 2013; 2013:675059. [PMID: 24024066 PMCID: PMC3759276 DOI: 10.1155/2013/675059] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/12/2013] [Accepted: 07/10/2013] [Indexed: 12/22/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) is a rare neoplasm described in several tissues and organs including genitourinary system, lung, head, and neck. The etiology of IMT is contentious, and whether it is a postinflammatory process or a true neoplasm remains controversial. To our knowledge, we report the first reported case of IMT of urinary bladder in a pediatric patient with Wolf-Hirschhorn (WHS). We also review the literature about patients with associated neoplasia.
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15
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Jenkins LC, Whittington E, Ciancio G, Jorda M. Inflammatory myofibroblastic tumor of the kidney. J Urol 2013; 189:2310-1. [PMID: 23507397 DOI: 10.1016/j.juro.2013.03.036] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 03/11/2013] [Indexed: 10/27/2022]
Affiliation(s)
- Lawrence C Jenkins
- Department of Urology, Transplant Institute, Jackson Memorial Hospital, Miami, Florida, USA
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Czerwinski M, Dave S. Pediatric renal inflammatory myofibroblastic tumours: A case report and review of the etiology and management options. Can Urol Assoc J 2012; 6:E150-3. [PMID: 23093568 DOI: 10.5489/cuaj.11120] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/19/2022]
Abstract
Inflammatory myofibroblastic tumours (IMTs) have been described in lung, bladder, spleen, breast, pancreas, liver, colon, spermatic cord, prostate, peripheral nerves, orbit and kidney. Traditionally believed as having a reactive pathogenesis, IMTs are now viewed more as a neoplasm. This report describes a case of a renal IMT in a 14-year-old girl with spina bifida associated neurogenic bladder and a history of recurrent urinary tract infections. This represents a unique case as pediatric renal IMTs are very rare in the literature. We discuss how this patient was managed and how she presented compared to other reported cases.
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17
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Huang YH, Zhong DJ, Tang J, Han JJ, Yu JD, Wang J, Wan YL. Inflammatory myofibroblastic tumor of the liver following renal transplantation. Ren Fail 2012; 34:789-91. [PMID: 22681584 DOI: 10.3109/0886022x.2012.673446] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/27/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT), previously named inflammatory pseudotumor, is a benign lesion, the exact etiology of which remains obscure; immunosuppression and infections have been speculated to be responsible for the development of pseudotumor. IMT associated with transplantation is rarely reported; we report the first case of IMT of the liver in a renal transplantation patient, who presented with symptoms of abdominal pain. The findings of computed tomography suggested hepatocellular carcinoma or liver abscess, and surgical resection was performed. The lesion was pathologically diagnosed as IMT.
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Affiliation(s)
- Yong-Heng Huang
- Department of Hepatobiliary Surgery, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou, Guangdong Province, PR China
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18
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Abu-Wasel B, Eltawil KM, Molinari M. Benign inflammatory pseudotumour mimicking extrahepatic bile duct cholangiocarcinoma in an adult man presenting with painless obstructive jaundice. BMJ Case Rep 2012; 2012:006514. [PMID: 22739336 DOI: 10.1136/bcr-2012-006514] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/19/2022] Open
Abstract
Inflammatory pseudotumours (IPTs) of the biliary tract are extremely rare and heterogeneous by aetiology and clinical presentation. They might cause biliary obstruction and mimic cholangiocarcinomas and their final diagnosis is usually achieved only after surgical excision. The most characteristic feature of IPT is the presence of chronic inflammatory cell infiltrates with variable degree of proliferating fibrous tissue. IPTs have the potential for recurrence even after resection and if untreated they can grow causing a variety of symptoms due to compression of the surrounding structures and organs. Despite the significant improvement of modern imaging techniques, preoperative distinction between IPTs of the biliary system and malignancies is extremely difficult. Histological diagnosis poses a clinical challenge because sampling is often suboptimal. Although rare, IPTs should be in the differential diagnosis of patients who present with painless jaundice and no other clinical symptoms or signs characteristics of cholangiocarcinomas.
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Affiliation(s)
- Bassam Abu-Wasel
- Department of Surgery, Dalhousie University, Halifax, Nova Scotia, Canada
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19
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Fragoso AC, Eloy C, Estevão-Costa J, Campos M, Farinha N, Lopes JM. Abdominal inflammatory myofibroblastic tumor a clinicopathologic study with reappraisal of biologic behavior. J Pediatr Surg 2011; 46:2076-82. [PMID: 22075336 DOI: 10.1016/j.jpedsurg.2011.07.009] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/09/2011] [Revised: 07/04/2011] [Accepted: 07/04/2011] [Indexed: 12/17/2022]
Abstract
BACKGROUND AND PURPOSE Inflammatory myofibroblastic tumor (IMT) is a proliferative lesion of controversial nosology and uncertain prognosis. In an attempt to acquire further understanding of pathogenesis and biologic behavior, we surveyed abdominal IMTs managed over the last 12 years at a single institution. METHODS Intra-abdominal IMTs treated between 1995 and 2007 were reviewed concerning demographic, clinical, and pathologic features as well as therapeutic management and outcome. All specimens were reevaluated by histologic examination and immunohistochemistry. RESULTS There were 7 patients (4 males; age range, 28 days to 14 years). Five lesions were located in alimentary tract: 1 gastric presenting with bleeding, 1 hepatic presenting with a thoracic wall mass, 1 pancreatic and 2 colonic presenting with obstructive symptoms. One splenic IMT was found incidentally. The remaining case arose from the adrenal gland and presented with a palpable mass. The gastric and adrenal IMTs had evidence of a previous or concomitant infectious setting. Five lesions were excised. The pancreatic IMT underwent a drainage procedure followed by steroid administration, and the hepatic lesion received antibiotics. Histopathology revealed characteristic findings of IMT. Expression of anaplastic lymphoma kinase was negative in all cases. At a median follow-up of 6 years (range, 3-15), all children were asymptomatic with no recurrences. The hepatic and pancreatic IMT displayed complete and near total regression, respectively. CONCLUSION A benign behavior of abdominal IMTs was observed even in patients not undergoing surgical excision. Although IMT remains a surgical disease, a conservative approach may be reasonable in select cases.
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Affiliation(s)
- Ana Catarina Fragoso
- Division of Pediatric Surgery, Faculty of Medicine, University of Porto, Hospital S. João, 4200-319 Porto, Portugal
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Abstract
Information is presented on the pathology of spindle cell sarcomas. Synovial sarcoma, malignant peripheral nerve sheath tumor, fibrosarcoma, inflammatory myofibroblastic tumor, low-grade myofibrosarcoma, leiomyosarcoma, spindle cell rhabdomyosarcoma, and endothelial neoplasms are discussed in terms of an overview of the tumor, microscopic and gross features, diagnostic techniques, genetic markers, differential diagnosis, clinical details, and prognosis.
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Affiliation(s)
- Cyril Fisher
- Department of Histopathology, The Royal Marsden Hospital, 203 Fulham Road, London SW3 6JJ, UK
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Pfeifer L, Agaimy A, Janka R, Boxberger F, Wein A, Neurath MF, Siebler J. Complete Long-Term Remission of an Inflammatory Pseudotumor under Corticosteroid Therapy. Case Rep Oncol 2011; 4:304-10. [PMID: 21734886 PMCID: PMC3124465 DOI: 10.1159/000329415] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/25/2022] Open
Abstract
Inflammatory pseudotumors (IPT) form a group of etiologically, histologically, and biologically heterogeneous tumefactive lesions that are histologically characterized by prominent inflammatory infiltrates. IPT has been described in various organs including the lungs, bladder, liver, spleen, heart, and others. It may mimic a malignant tumor clinically and radiologically. We report a case of a 26-year-old woman with an ALK1-negative IPT (7 cm in maximal diameter) mainly located in the 12th right back muscles, surrounding a fractured rib. Histologically, the tumor consisted of an inflammatory infiltrate composed predominantly of diffusely distributed lymphoplasmacytic cells and stromal fibroblasts associated with focal obliterative phlebitis. Conservative steroid treatment resulted in complete remission and the patient remained disease-free for more than 1 year later. To our knowledge this is the first report of IPT involving the skeletal back muscle and complete resolution under corticosteroid treatment.
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Affiliation(s)
- Lukas Pfeifer
- Department of Internal Medicine 1, University of Erlangen, Erlangen, Germany
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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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25
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Alaggio R, Cecchetto G, Bisogno G, Gambini C, Calabrò ML, Inserra A, Boldrini R, De Salvo GL, G d'Amore ES, Dall'igna P. Inflammatory myofibroblastic tumors in childhood: a report from the Italian Cooperative Group studies. Cancer 2010; 116:216-26. [PMID: 19852031 DOI: 10.1002/cncr.24684] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Abstract
BACKGROUND Inflammatory myofibroblastic tumors (IMTs) are myofibroblastic lesions with unpredictable biologic behavior that occur at a young age. For this report, the authors investigated clinicopathologic features in a series of pediatric IMTs. The objective of the study was to identify morphologic or immunohistochemical prognostic markers and the possible pathogenic role of human herpes virus 8 (HHV-8). METHODS Twenty-six patients were observed over a period of 18 years. Clinical/histologic data were reviewed, and immunohistochemical/molecular studies were performed. RESULTS Patients ages 8-216 months (median age, 60 months) presented with tumors of the lung-bronchus (8 patients), abdomen (17 patients), and thoracic wall (1 patient). Twenty-one patients underwent complete excision, and microscopic or macroscopic residual disease was present in 5 of those patients. Chemotherapy was received by 5 patients. After a median follow-up of 6.6 years, 24 patients were in complete remission, and 2 patients had died of disease. Local recurrences were observed in 6 patients (including 4 recurrences that occurred after a complete excision). Cytologic atypia, low inflammatory infiltrate, and a rich myxoid pattern were detected in patients who had recurrent disease or a poor prognosis. Anaplastic lymphoma kinase (ALK) was positive in 7 patients (including 2 patients with recurrent disease). No correlation between clusterin expression and prognosis was demonstrated. HHV-8 was identified in 1 pulmonary IMT. CONCLUSIONS IMTs are locally aggressive lesions. In this series, the local recurrence rate was 23%, and the 5-year and 10-year event-free survival rates were 87.4% and 72.8%, respectively. The results indicated that the treatment of choice is a complete, nonmutilating excision; chemotherapy may be given to patients who have microscopic or macroscopic residual disease, although the results are controversial; cytologic atypia and positive ALK status are more frequent in aggressive tumors, whereas metastatic tumors are negative for ALK; and HHV8 is not involved in the pathogenesis of IMT.
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Affiliation(s)
- Rita Alaggio
- Pathology Department, University Hospital of Padova, Padova, Italy.
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Case report: inflammatory myofibroblastic tumor of the duodenum. J Gastrointest Cancer 2009; 39:79-81. [PMID: 19142589 DOI: 10.1007/s12029-008-9042-y] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/07/2008] [Accepted: 12/10/2008] [Indexed: 10/21/2022]
Abstract
INTRODUCTION We report a very rare case of inflammatory myofibroblastic tumor of the duodenum. A 16-year-old boy underwent esophagogastroduodenoscopy for intermittent epigastric pain, night sweats, and malaise. CASE REPORT An exophytic mass lesion was found in the first part of the duodenum, but biopsies were non-diagnostic. Computed tomography confirmed a 60-mm mass with no lymphadenopathy, and the patient underwent surgical resection. Histology revealed spindle cells with the morphological and immunophenotypical profile of myofibroblasts on a background of mixed inflammatory infiltrate, typical of inflammatory myofibroblastic tumor. Six months after surgery, the patient developed a recurrence, and this was successfully treated by immunosuppression. Currently, the patient is asymptomatic, and there is no radiological or pathological evidence of disease.
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28
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Pelosi G, Sonzogni A, De Pas T, Galetta D, Veronesi G, Spaggiari L, Manzotti M, Fumagalli C, Bresaola E, Nappi O, Viale G, Rosai J. Review article: pulmonary sarcomatoid carcinomas: a practical overview. Int J Surg Pathol 2009; 18:103-20. [PMID: 19124452 DOI: 10.1177/1066896908330049] [Citation(s) in RCA: 111] [Impact Index Per Article: 6.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/16/2022]
Abstract
Pulmonary sarcomatoid carcinomas (PSCs) are currently defined as poorly differentiated non-small-cell carcinomas containing a component with sarcoma or sarcoma-like (spindle and/or giant cell) features. They consist of 5 major histological variants, namely pleomorphic carcinoma, spindle cell carcinoma, giant cell carcinoma, carcinosarcoma, and pulmonary blastoma. The segregation of PSCs into a distinct clinicopathologic entity seems justified on the basis of morphologic, behavioral, and genotypic/phenotypic attributes. As a group, PSCs generally run an aggressive clinical course and may cause major difficulties in the differential diagnosis with other primary and secondary malignancies of the lung. At present, PSCs are believed to represent a family of carcinomas "in transition," in which diverse pathways of clonal evolution account for histological differences of a common ancestor lesion. The sarcomatous or sarcomatoid component of these tumors is thought to derive from carcinoma cells during the progression of carcinogenesis through the activation of an epithelial-mesenchymal transition program leading to sarcomatous transformation or metaplasia (conversion paradigm). Conceivably, targeting the epithelial-mesenchymal transition program could become a valid therapeutic strategy for these life-threatening tumors, whose sensitivity to current medical manipulation is disappointing.
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Affiliation(s)
- Giuseppe Pelosi
- Division of Pathology and Laboratory Medicine, University of Milan School of Medicine, Milan.
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29
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Nistal M, Gonzalez-Peramato P, Serrano A, Reyes-Mugica M, Cajaiba MM. Primary intratesticular spindle cell tumors: interdigitating dendritic cell tumor and inflammatory myofibroblastic tumor. Int J Surg Pathol 2008; 19:104-9. [PMID: 18805870 DOI: 10.1177/1066896908323505] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
Spindle cell neoplasms arising in the testis are uncommon; most cases belong to the category of gonadal stromal tumors, and the presence of distinctive clinical and pathological features usually lead to a definitive diagnosis. In some instances, however, the diagnosis of these tumors can be challenging and special techniques are needed. The present study reports 2 unusual cases of primary intratesticular tumors showing a striking morphology, characterized by spindle to stellate cells in a lymphoid background. The diagnosis of interdigitating dendritic cell tumor and inflammatory myofibroblastic tumor was established in the 2 cases, and their probable origin in cells of the accessory immune system is discussed. Although both tumors share similar histological features, their immunohistochemical profiles were decisive for a definitive diagnosis.
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Affiliation(s)
- Manuel Nistal
- Department of Pathology, Hospital La Paz, Madrid, Spain
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Bedke J, Buse S, Esposito I, Schirmacher P, Haferkamp A, Hohenfellner M. Evaluation and management of a patient with a bladder mass of uncertain etiology. NATURE CLINICAL PRACTICE. UROLOGY 2008; 5:509-514. [PMID: 18695714 DOI: 10.1038/ncpuro1172] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 10/28/2007] [Accepted: 06/02/2008] [Indexed: 05/26/2023]
Abstract
BACKGROUND A healthy, parous, nonsmoking, 36-year-old woman consulted her gynecologist for nonspecific bladder pain. Urinary tract infection was ruled out. Vaginal ultrasonography and MRI revealed an undefined tumor between the bladder and the uterus. The patient refused further testing until tumor growth was detected at a scheduled appointment 5 months after presentation. She was referred to a urology department at this time. INVESTIGATIONS Physical examination, urine culture, medical history, cystoscopy, MRI, angiography, intraoperative frozen section analysis, and final histology. DIAGNOSIS Inflammatory myofibroblastic tumor of the bladder. Management Partial cystectomy with complete excision of the tumor from the trigonal and posterior wall of the bladder by median laparotomy.
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Affiliation(s)
- Jens Bedke
- Department of Urology, Eberhard-Karls-University Tübingen, Tübingen, Germany.
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31
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Cytokeratin-positive interstitial reticulum cell tumors of lymph nodes: a case report and review of literature. Chin Med J (Engl) 2008. [DOI: 10.1097/00029330-200804010-00016] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/30/2022] Open
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Saint-Blancard P, Harket A, Tine I, Daumas-Duport C, Richard de Soultrait F. Une lésion rare du système nerveux central : la pseudotumeur inflammatoire. Neurochirurgie 2008; 54:37-40. [DOI: 10.1016/j.neuchi.2008.01.001] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/21/2007] [Accepted: 12/05/2007] [Indexed: 12/12/2022]
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Ma Y, Zieske AW, Fenves AZ. Bilateral infiltrating renal inflammatory pseudotumor responsive to corticosteroid therapy. Am J Kidney Dis 2008; 51:116-20. [PMID: 18155540 DOI: 10.1053/j.ajkd.2007.08.028] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/28/2007] [Accepted: 08/07/2007] [Indexed: 01/17/2023]
Abstract
Inflammatory pseudotumor (IPT) is a quasi-neoplastic lesion that most commonly involves the lung, but has been shown to occur in nearly every tissue type. Renal involvement is very uncommon. We report the second case of IPT ever published presenting as bilateral infiltrating renal masses. Although most renal IPTs were treated with nephrectomy, our patient was managed successfully with conservative steroid treatment, thereby avoiding the alternative of dialysis or kidney transplantation.
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Affiliation(s)
- Yanjun Ma
- Department of Internal Medicine, Baylor University Medical Center, Dallas, TX 75246, USA.
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Abstract
Inflammatory pseudotumour is a generic term applied to a variety of neoplastic and non-neoplastic entities that share a common histological appearance, namely a cytologically bland spindle cell proliferation with a prominent, usually chronic inflammatory infiltrate. Over the last two decades, inflammatory myofibroblastic tumour (IMT) has emerged from within the broad category of inflammatory pseudotumour, with distinctive clinical, pathological and molecular features. IMT shows a predilection for the visceral soft tissues of children and adolescents and has a tendency for local recurrence, but only a small risk of distant metastasis. Characteristic histological patterns include the fasciitis-like, compact spindle cell and hypocellular fibrous patterns, which are often seen in combination within the same tumour. Chromosomal translocations leading to activation of the ALK tyrosine kinase can be detected in approximately 50% of IMTs, particularly those arising in young patients. This review will examine the clinical, pathological, and molecular genetic features of IMT and discuss an approach to diagnosis and differential diagnosis.
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Affiliation(s)
- B C Gleason
- Department of Pathology, Brigham and Women's Hospital and Harvard Medical School, Boston, MA 02115, USA
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35
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Abstract
"Inflammatory pseudotumors" (IPTs) embrace a heterogeneous spectrum of reactive, infective, and neoplastic entities, that are characterized by a clinical mass composed of a histologic proliferation of spindle cells in a background of inflammatory cells and collagen fibers. Although a spectrum of microorganisms have been identified in infective IPTs, mycobacterial infective IPTs are reported most commonly. We document 5 solitary cryptococcal IPTs, in 2 males and 3 females, aged 19 to 43 years, in the soft tissues of the anterior chest wall, thigh, and arm. All were HIV-positive and had been treated for disseminated cutaneous and/or meningeal cryptococcosis with antifungal therapy, 6 to 12 months earlier. The specimens demonstrated a storiform arrangement of plump spindle cells, in addition to spindle and polygonal cells that were arranged in a haphazard manner. Background lymphocytes, plasma cells, and fibrosis were noted, in addition to scattered giant cells and focal necrosis. On high-power examination, Cryptococcus neoformans yeasts were identified within and between vacuolated spindle and polygonal cells on routine and special stains, confirming cryptococcal IPTs. Immunophenotyping of the spindle cells confirmed a mixed histiocytic and myofibroblastic lineage, with a predominance of the former. In documenting 5, hitherto unreported, pseudotumoral spindle cell reactions to C. neoformans, we not only highlight the need for intense appraisal of all IPTs for infective agents on routine and special stains and investigations, but also postulate that a complex host-fungus interaction, coupled with an exuberant, myofibroblastic response to incomplete therapy, are the pathogenetic drive for the pseudotumoral presentation.
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Affiliation(s)
- Yetish Sing
- Department of Anatomical Pathology, Nelson R Mandela School of Medicine, University of KwaZulu Natal and National Health Laboratory Service, Durban, KwaZulu Natal, South Africa
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36
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Díaz-Torné C, Narváez J, De Lama E, Diez-García M, Narváez JA, Bernad B, Llatjos R, Nolla JM, Valverde J. Inflammatory pseudotumor of the liver associated with rheumatoid arthritis. ACTA ACUST UNITED AC 2007; 57:1102-6. [PMID: 17665472 DOI: 10.1002/art.22904] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/17/2022]
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Megremis S, Papamitsaki E, Ieromonachou P, Zois E. Inflammatory myofibroblastic tumor of the paratestis: sonographic appearance with pathologic correlation. JOURNAL OF ULTRASOUND IN MEDICINE : OFFICIAL JOURNAL OF THE AMERICAN INSTITUTE OF ULTRASOUND IN MEDICINE 2007; 26:1227-30. [PMID: 17715318 DOI: 10.7863/jum.2007.26.9.1227] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 05/16/2023]
Affiliation(s)
- Stylianos Megremis
- First Department of Radiology, Venizelio General Hospital, Iraklio, Crete, Greece.
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38
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Saleem MI, Ben-Hamida MA, Barrett AM, Bunn SK, Huntley L, Wood KM, Yelbuz TM. Lower abdominal inflammatory myofibroblastic tumor -an unusual presentation- a case report and brief literature review. Eur J Pediatr 2007; 166:679-83. [PMID: 17109166 DOI: 10.1007/s00431-006-0305-y] [Citation(s) in RCA: 23] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/08/2006] [Accepted: 08/31/2006] [Indexed: 12/17/2022]
Abstract
A 9-year-old girl presented with lethargy, malaise & chest pain. Her blood counts confirmed hypochromic microcytic anemia. She was prescribed iron supplements. Subsequently she was admitted to our hospital with fever and increasing chest and abdominal pain. She was treated with antibiotics, and a diagnosis of "early chest infection" was made. Over the following 2 weeks she failed to improve, and her anemia worsened. She was readmitted, and found to have a mass in her lower abdomen with pressure symptoms on her bowel and bladder. A white-cell scan showed increased uptake in right lower quadrant. An ultrasound and a CT scan confirmed a mass adjacent to her bladder. Needle biopsy showed it to be an unusual localization of an inflammatory myofibroblastic tumor (IMT) of cecum. A presentation with chest pain, fever, anemia and pressure symptoms was highly unusual of a lower abdominal IMT mass. She had a successful excision of the tumor, with resolution of her symptoms.
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Affiliation(s)
- M Ilyas Saleem
- Department of Pediatrics, West Cumberland Hospital, Hensingham, Whitehaven, Cumbria, UK.
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39
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Sukov WR, Cheville JC, Carlson AW, Shearer BM, Piatigorsky EJ, Grogg KL, Sebo TJ, Sinnwell JP, Ketterling RP. Utility of ALK-1 protein expression and ALK rearrangements in distinguishing inflammatory myofibroblastic tumor from malignant spindle cell lesions of the urinary bladder. Mod Pathol 2007; 20:592-603. [PMID: 17396140 DOI: 10.1038/modpathol.3800776] [Citation(s) in RCA: 75] [Impact Index Per Article: 4.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/22/2023]
Abstract
Inflammatory myofibroblastic tumor of the urinary bladder is an unusual spindle cell neoplasm that displays cytologic atypia, infiltrative growth and mitotic activity mimicking malignant tumors, such as leiomyosarcoma, rhabdomyosarcoma and sarcomatoid carcinoma. The objective of this study was to determine if anaplastic lymphoma kinase (ALK-1) protein expression detected by immunohistochemistry and ALK rearrangements detected by fluorescence in situ hybridization (FISH) were useful in distinguishing inflammatory myofibroblastic tumor from malignant spindle cell tumors of the urinary bladder. In inflammatory myofibroblastic tumor, ALK-1 expression was identified in 13 of 21 cases (62%) and ALK rearrangements in 14 of 21 cases (67%). All cases of inflammatory myofibroblastic tumor demonstrating ALK-1 expression, carried ALK rearrangements. One case negative for ALK-1 expression exhibited ALK rearrangement. ALK rearrangements were more common in women (P=0.0032). Leiomyosarcoma, sarcomatoid carcinoma, embryonal rhabdomyosarcoma and reactive myofibroblastic proliferations were negative for ALK-1 protein and ALK rearrangements. Immunohistochemistry using markers of muscle, epithelial, neural, and follicular dendritic cell differentiation showed overlap between inflammatory myofibroblastic tumor with and without ALK gene rearrangements, and between inflammatory myofibroblastic tumor and spindle cell malignancies. However, coexpression of cytokeratin and muscle-specific antigens was unique to inflammatory myofibroblastic tumor, observed in approximately half the tumors. This study indicates that detection of ALK protein and ALK gene rearrangements are useful in distinguishing inflammatory myofibroblastic tumor from spindle cell malignancies in the urinary bladder. Additionally, our findings suggest that ALK rearrangement is the primary mechanism for ALK activation and that inflammatory myofibroblastic tumor likely represents a heterogeneous group of spindle cell proliferations with the majority associated with ALK translocations, and the remaining associated with other etiologies.
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Affiliation(s)
- William R Sukov
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN 55902, USA
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40
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Farris AB, Mark EJ, Kradin RL. Pulmonary “inflammatory myofibroblastic” tumors: a critical examination of the diagnostic category based on quantitative immunohistochemical analysis. Virchows Arch 2007; 450:585-90. [PMID: 17372757 DOI: 10.1007/s00428-007-0395-6] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/18/2006] [Revised: 02/22/2007] [Accepted: 02/23/2007] [Indexed: 10/23/2022]
Abstract
The World Health Organization classification applies the term "pulmonary inflammatory myofibroblastic tumor" to a histologically variegate set of pulmonary inflammatory pseudotumors. However, often these lesions bear little resemblance to tumors of myofibroblastic origin. To elucidate histogenesis, we examined 18 cases from our institution files. The cases were stained with antibodies to smooth muscle actin (SMA), Factor XIIIa, CD3, CD20, CD68, S-100, anaplastic lymphoma kinase (ALK-1), and human herpevirus-8 (HHV-8). The percentage of positive-staining cells within a defined tumor area (400,000 microm(2)) was determined by light microscopy and morphometric analysis. Ten cases (56%) showed myofibroblastic differentiation, as judged by positive SMA staining of spindle cells. All cases showed substantial numbers of CD68+, Factor XIIIa+, and S-100+ monocytoid cells. Fifty percent were ALK-1+, and one was HHV-8+. We conclude that the term "inflammatory myofibroblastic tumor" is a misnomer, as nearly half of cases show no myofibroblastic differentiation. Instead, the results suggest that these lesions are composed predominantly of cells of macrophage-dendritic cell lineage. Although the multiplicity of terms previously applied to these lesions is cumbersome, retaining a descriptive phenomenological terminology may ultimately promote accurate elucidation of pathogenesis.
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Affiliation(s)
- A B Farris
- Department of Pathology, Massachusetts General Hospital, Boston, MA 02114, USA
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41
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Montgomery EA, Shuster DD, Burkart AL, Esteban JM, Sgrignoli A, Elwood L, Vaughn DJ, Griffin CA, Epstein JI. Inflammatory myofibroblastic tumors of the urinary tract: a clinicopathologic study of 46 cases, including a malignant example inflammatory fibrosarcoma and a subset associated with high-grade urothelial carcinoma. Am J Surg Pathol 2007; 30:1502-12. [PMID: 17122505 DOI: 10.1097/01.pas.0000213280.35413.1b] [Citation(s) in RCA: 146] [Impact Index Per Article: 8.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/12/2023]
Abstract
Inflammatory myofibroblastic tumor (IMT) of the urinary tract, also termed postoperative spindle cell nodule, inflammatory pseudotumor, and pseudosarcomatous fibromyxoid tumor, is rare and in the past was believed to reflect diverse entities. We reviewed a series of 46 IMTs arising in the ureter, bladder, and prostate, derived primarily from a large consultation practice. There were 30 male and 16 females aged 3 to 89 years (mean 53.6). Lesions were 1.2 to 12 cm (mean 4.2). There was a history of recent prior instrumentation in 8 cases. Morphology was similar to that previously described for IMT occurring in this region, with the exception of 1 case that focally appeared sarcomatous. Polypoid cystitis coexisted in 5 patients (11%). Mitoses were typically scant (0 to 20/10 hpf, mean 1). Necrosis was seen in 14 (30%) cases. Invasion of the muscularis propria was documented in 19 (41%). By immunohistochemistry (IHC), lesions at least focally expressed anaplastic lymphoma kinase (ALK) (20/35, 57%), AE1/3 (25/34, 73%), CAM5.2 (10/15, 67%), CK18 (6/6, 100%), actin (23/25, 92%), desmin (15/19, 79%), calponin (6/7, 86%), caldesmon (4/7, 57%, rare cells), p53 (10/13, 77%), and most lacked S100 (0/14), CD34 (0/13), CD117 (2/13, 15%), CD21 (0/5), and CD23 (0/3). ALK gene alterations were detected by fluorescence in situ hybridization (FISH) in 13/18 (72%) tested cases, including 2 with prior instrumentation; 13/18 (72%) showed agreement between FISH ALK results and ALK protein results by IHC. Most bladder IMTs were managed locally, but partial cystectomy was performed as the initial management in 7 cases and cystectomy in 1 (1 IMT was initially misinterpreted as carcinoma, 1 IMT was found incidentally as a separate lesion in a cystectomy specimen performed for urothelial carcinoma). Follow-up was available in 32 cases (range 3 to 120 mo; mean 33; median 24). There were 10 patients with recurrences (2 with 2 recurrences). Recurrences were unassociated with muscle invasion or with ALK alterations. In 2 cases, tumors of the urinary tract (TURs) showing IMT preceded (1 and 2 mo, respectively) TURs showing sarcomatoid carcinoma with high-grade invasive urothelial carcinoma accompanied with separate fragments of IMT. Even on re-review the IMT in these 2 cases were morphologically indistinguishable from other cases of IMT, with FISH demonstrating ALK alterations in the IMT areas in one of them. Both these patients died of their carcinomas. Lastly, there was 1 tumor with many morphological features of IMT and an ALK rearrangement, yet overtly sarcomatous. This case arose postirradiation for prostate cancer 4 years before the development of the lesion, with tumor recurrence at 4 months and death from intra-abdominal metastatic disease at 9 months. In summary, urinary tract IMTs are rare and share many features with counterparts in other sites, displaying similar morphology and immunogenotypic features whether de novo or postinstrumentation. Typical IMTs can be locally aggressive, sometimes requiring radical surgical resection, but none of our typical cases metastasized, although they can rarely arise contemporaneously with sarcomatoid urothelial carcinomas. For these reasons, close follow-up is warranted.
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MESH Headings
- Adolescent
- Adult
- Aged
- Aged, 80 and over
- Anaplastic Lymphoma Kinase
- Biomarkers, Tumor/metabolism
- Carcinoma, Transitional Cell/enzymology
- Carcinoma, Transitional Cell/genetics
- Carcinoma, Transitional Cell/pathology
- Child
- Child, Preschool
- Female
- Fibrosarcoma/enzymology
- Fibrosarcoma/genetics
- Fibrosarcoma/pathology
- Granuloma, Plasma Cell/enzymology
- Granuloma, Plasma Cell/genetics
- Granuloma, Plasma Cell/pathology
- Humans
- In Situ Hybridization, Fluorescence
- Inflammation/pathology
- Male
- Middle Aged
- Prostate/enzymology
- Prostate/pathology
- Protein-Tyrosine Kinases/genetics
- Protein-Tyrosine Kinases/metabolism
- Receptor Protein-Tyrosine Kinases
- Ureter/enzymology
- Ureter/pathology
- Urinary Bladder/enzymology
- Urinary Bladder/pathology
- Urologic Diseases/enzymology
- Urologic Diseases/genetics
- Urologic Diseases/pathology
- Urothelium/enzymology
- Urothelium/pathology
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42
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Farris AB, Kradin RL. Follicular localization of dendritic cells in a xanthomatous inflammatory tumor of lung associated with human herpes virus-8 infection. Virchows Arch 2006; 449:726-9. [PMID: 17106709 DOI: 10.1007/s00428-006-0318-y] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/24/2006] [Accepted: 09/15/2006] [Indexed: 11/30/2022]
Abstract
A 17-year-old man was treated with chemotherapy and radiation for nodular sclerosing Hodgkin lymphoma that presented as a left chest wall mass. Ten years later, a left upper lobe lung tumor was identified. The tumor resection demonstrated a 1.3-cm yellow lung nodule composed of epithelioid and spindled lipid-laden CD68+ and Factor XIIIa+ macrophages. Distinct follicular structures with dendritic cells positive for CD1a, fascin, and ALK-1 and largely devoid of intracytoplasmic lipid were a distinguishing feature of the lesion. Most of the xanthomatous macrophages expressed human herpes virus-8 antigen. The current World Health Organization classification of "inflammatory myofibroblastic tumors" is examined, and the association of a subset of "inflammatory pseudotumors" with immunodeficiency states and opportunistic infection is discussed.
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Affiliation(s)
- A B Farris
- Department of Pathology, Massachusetts General Hospital, Boston, MA 02114, USA
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43
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Shia J, Chen W, Tang LH, Carlson DL, Qin J, Guillem JG, Nobrega J, Wong WD, Klimstra DS. Extranodal follicular dendritic cell sarcoma: clinical, pathologic, and histogenetic characteristics of an underrecognized disease entity. Virchows Arch 2006; 449:148-58. [PMID: 16758173 DOI: 10.1007/s00428-006-0231-4] [Citation(s) in RCA: 71] [Impact Index Per Article: 3.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/24/2006] [Accepted: 05/05/2006] [Indexed: 12/19/2022]
Abstract
It has been more than 10 years since follicular dendritic cell (FDC) sarcoma was first reported to occur in extranodal sites, yet extranodal FDC sarcoma still appears underrecognized, and its clinical and pathological characteristics remain to be defined. This study analyzed the clinical and pathological findings of three such cases that the authors encountered recently and 43 previously reported cases identified in the literature. Assessment of all 46 cases showed a slight female predominance (1.2:1) with a median age of 41.5 years. One-third of the cases were misdiagnosed at initial evaluation mainly because the possibility of FDC sarcoma was not considered. When considered, this disease had distinct pathological characteristics that allowed an accurate diagnosis. Staining for FDC markers, CD21, CD35, and clusterin was particularly helpful. The pathogenesis of the disease appeared heterogeneous, and associated factors included Epstein-Barr virus infection (in hepatic cases) and inflammatory pseudotumor-like conditions. Treatment modality varied widely although surgical resection was often included. With a median follow-up of 18 months, 43% of the cases recurred and 7% died of disease. The 5-year recurrence-free survival was 27.4%. From data available at the current time, we were not able to identify prognostically significant pathologic factors.
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Affiliation(s)
- Jinru Shia
- Department of Pathology, Memorial Sloan-Kettering Cancer Center, 1275 York Avenue, New York, NY 10021, USA
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44
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Agaimy A, Wünsch PH. Follicular dendritic cell tumor of the gastrointestinal tract: Report of a rare neoplasm and literature review. Pathol Res Pract 2006; 202:541-8. [PMID: 16564140 DOI: 10.1016/j.prp.2006.01.013] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/15/2005] [Accepted: 01/30/2006] [Indexed: 01/09/2023]
Abstract
Follicular dendritic cell (FDC) tumor is a rare neoplasm of the accessory immune system showing FDC differentiation. Histologically, a conventional type and an inflammatory pseudotumor (IPT-like) type are recognized. The etiology of FDC tumor is unknown. While rare FDC tumors were associated with hyaline-vascular Castleman's disease (HVCD), hepatosplenic IPT-like FDC tumors consistently harbor EBV infection. FDC tumors of the gastrointestinal (GI) tract and mesentery/omentum are exceedingly rare, with only 17 cases reported so far. We report an additional case of an IPT-like FDC tumor of the ileum and mesentery in a 52-year-old schizophrenic man. The tumor consisted of highly atypical multinucleated giant cells in a background of intense lymphoid infiltrate with prominent eosinophilia reminiscent of Hodgkin's lymphoma. Tumor cells were immunoreactive for vimentin, CD21, CD35, fascin, smooth muscle actin and CD68, but were negative for all lineage-specific lymphoreticular, myeloid, mesenchymal and epithelial markers. Immunostaining for HHV-8 and in situ hybridization for EBV-encoded RNA (EBER) were negative. Some mesenteric lymph nodes showed HVCD-like changes. The differential diagnostic considerations of this unusual and rare neoplasm, mainly lymphocyte-rich GI stromal tumor (GIST), malignant lymphoma and inflammatory neoplasia of diverse histogenetic types, will be discussed together with a literature review on gastrointestinal FDC tumors.
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Affiliation(s)
- Abbas Agaimy
- Institut für Pathologie, Klinikum Nürnberg, Prof.-Ernst-Nathan-Strasse 1, 90419 Nürnberg, Germany.
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