Inflammatory myofibrohistiocytic proliferation simulating sarcoma in children

Tang, Thomas T.; Segura, Annette D.; Oechler, Herbert W.; Harb, Joseph M.; Adair, Stuart; Gregg, David C.; Camitta, Bruce M.; Franciosi, Ralph A.

doi:10.1002/1097-0142(19900401)65:7<1626::aid-cncr2820650729>3.0.co;2-v

Cited by 128 publications

(73 citation statements)

References 25 publications

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“…IMT and IgG4SD are both fibro-inflammatory proliferations that are included in the larger constellation 6,7 Inflammatory myofibroblastic tumor originates anywhere in the body, but has a predilection for the abdominal cavity, pelvis, retroperitoneum, lungs, mediastinum, and upper respiratory tract. [14][15][16][17]24 In contrast to IMT, IgG4SD is an inflammatory disorder that is accompanied by an elevated serum IgG4 and is histologically characterized by an infiltrative inflammatory fibro-sclerosing process that usually contains abundant IgG4 positive plasma cells.…”

Section: Discussionmentioning

confidence: 99%

“…6 A reported 15-30% of patients have a clinical and laboratory syndrome of fever, malaise, weight loss, anemia, elevated erythrocyte sedimentation rate, thrombocytosis, polyclonal hypergammaglobulinemia, and other inflammatory marker elevations. [7][8][9][10][11][12][13] IgG4-related sclerosing disease (IgG4SD) is a steroid-responsive multiorgan system disorder that encompasses a spectrum of clinical conditions, includ ing autoimmune pancreatitis, sclerosing cholangitis, chronic sclerosing sialadenitis, tubulointerstitial nephritis, cutaneous pseudolymphoma, and sclerosing processes in the lung and pleura, liver, breast, and meninges. 5,[14][15][16][17] The histological findings of fibrosis and chronic inflammation are relatively nonspecific, and there are no known unique genetic markers.…”

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confidence: 99%

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IgG4 plasma cells in inflammatory myofibroblastic tumor: inflammatory marker or pathogenic link?

et al. 2011

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Inflammatory myofibroblastic tumor is a rare mesenchymal neoplasm that harbors an anaplastic lymphoma kinase (ALK) gene rearrangement in the majority of cases. It is composed of fibroblastic-myofibroblastic cells with a characteristic inflammatory infiltrate that consists predominantly of plasma cells. In contrast, IgG4-related sclerosing disease is a recently described multisystem disorder with a histological appearance similar to inflammatory myofibroblastic tumor. The plasma cell infiltrate is characteristic in IgG4-related sclerosing disease and has been studied as a tool to render this diagnosis. Histologically, the two disorders overlap, although there are significant clinical differences. This study analyzes the histological appearance of 36 inflammatory myofibroblastic tumors, compares them with IgG4-related sclerosing disease, and assesses the plasma cell profile using immunohistochemistry to determine the range and proportion of IgG4 plasma cells. The majority of patients were children and young adults, mainly with solitary masses and no clinical manifestations of IgG4-related sclerosing disease. ALK-1 positivity was present in 23 cases (64%). None showed obliterative phlebitis or prominent lymphoid aggregates. Of 36 inflammatory myofibroblastic tumors, 15 cases showed an IgG4/IgG ratio Z0.10, a cutoff described in the literature as supportive of IgG4-related sclerosing disease and up to 33 IgG4-positive plasma cells per high-power field indicating a mild-to-moderate increase as compared with IgG4-related sclerosing disease. Currently, the diagnostic recognition of inflammatory myofibroblastic tumor is based on clinicopathological features and diagnostic adjuncts, such as ALK-1 reactivity and genetic tests. Although inflammatory myofibroblastic tumor and IgG4-related sclerosing disease are distinct entities, a subset of inflammatory myofibroblastic tumors exhibit an IgG4/IgG ratio that is within the range for IgG4-related sclerosing disease. Therefore, the ratio alone cannot be used as a reliable discriminator between these two entities and other clinical and pathologic features must always be taken into account.

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Section: Discussionmentioning

confidence: 99%

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confidence: 99%

IgG4 plasma cells in inflammatory myofibroblastic tumor: inflammatory marker or pathogenic link?

et al. 2011

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“…Clinical and pathological findings from a review of 13 cases of gastric IMT in children that has been reported under a variety of additional descriptive terms are summarized in Table 1 (4,(8)(9)(10)(11)(12)(13)(14)(15)(16). Most cases were of less than 6-yr-old children (92.3%) and females (69.2%).…”

Section: Discussionmentioning

confidence: 99%

Fever of Unknown Origin as a Presentation of Gastric Inflammatory Myofibroblastic Tumor in a Two-Year-Old Boy

et al. 2002

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Gastric inflammatory myofibroblastic tumor (IMT) is an extremely rare lesion with mimicking malignant features and accompanied with various clinical manifestations. Here we present a 2-yr-old boy who had a gastric IMT with a huge extragastric mass, which closely resembled a neuroblastoma on imaging studies. He experienced intermittent fever and poor appetite for 6 weeks. Fever remained up to 38°C even on the operation day. He underwent partial gastrectomy and distal pancreatectomy with splenectomy including the tumor. The preoperative fever disappeared and did not recur in the postoperative course.

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“…[3][4][5] These terms are often confused with soft tissue sarcoma because of its recurrence tendency and local invasion. 5 Main stay of treatment is surgical resection and in some corticosteroids or NSAIDS may be useful. 6 …”

Section: Introductionmentioning

confidence: 99%

Jejunal inflammatory myofibroblastic tumor: a rare entity

2017

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Inflammatory myofibroblastic tumor is one of the rare solid tumor occurring in children. Main stay of treatment is surgical resection and in some corticosteroids or NSAIDS may be useful. Here, a case of 3-year-old female with inflammatory myofibroblastic tumor of jejunum is presented, that was evaluated clinically, investigated radiologically and finally histopathology confirmed the diagnosis. No complications occurred at peri and postoperative period. The patient was on regular follow-up and no recurrence had been documented yet in 1 year of follow up. In this article, we reviewed the literature for inflammatory myofibroblastic tumor.

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Inflammatory myofibrohistiocytic proliferation simulating sarcoma in children

Cited by 128 publications

References 25 publications

IgG4 plasma cells in inflammatory myofibroblastic tumor: inflammatory marker or pathogenic link?

IgG4 plasma cells in inflammatory myofibroblastic tumor: inflammatory marker or pathogenic link?

Fever of Unknown Origin as a Presentation of Gastric Inflammatory Myofibroblastic Tumor in a Two-Year-Old Boy

Jejunal inflammatory myofibroblastic tumor: a rare entity

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