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      Primary Inflammatory Myofibroblastic Tumors of the Breast with Metastasis: Radiographic and Histopathologic Predictive Factors

      case-report

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          Abstract

          Primary inflammatory myofibroblastic tumors (IMTs) of the breast are uncommon and metastasis of IMTs is extremely rare. To date, the natural course of this disease is not fully understood. Although patients with IMTs should undergo regular follow-up after complete surgical resection of the tumor, the appropriate interval and method of follow-up are unclear. We report the case of a patient with an IMT of the breast that metastasized 2 years after complete surgical resection. This unusual case emphasizes the importance of preoperative examinations to determine whether the IMT has atypical features that should guide the interval and method of follow-up.

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          Most cited references12

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          Comparison of DNA ploidy, histologic, and immunohistochemical findings with clinical outcome in inflammatory myofibroblastic tumors.

          Inflammatory myofibroblastic tumors (IMTs) are uncommon spindle cell proliferations that occur in the viscera and soft tissue of children and young adults. Their biologic potential is indeterminate: 25% of IMTs recur, and rare examples undergo malignant transformation (MT). This study investigates histologic features, DNA ploidy, and expression of apoptotic regulatory and oncogenic proteins in IMTs in an attempt to identify those deviances that might correlate with aggressive behavior or MT. Twenty-four formalin-fixed, paraffin-embedded IMTs for which clinical outcome was known were evaluated for cellularity, cytologic atypia, mitoses, ganglion-like cells, inflammatory infiltrate, DNA ploidy by flow cytometric examination, and bax, bcl-2, p53, and c-myc expression by immunohistochemical analysis. Sixteen (67%) of the IMTs did not recur, 6 (25%) recurred, and 2 (8%) underwent MT. Cellular atypia was observed in 69% of the cases without recurrence, 100% with recurrence, and 100% with MT. Ganglion-like cells were present in 56, 100, and 100% of the IMTs without recurrence, with recurrence, and with MT, respectively. There was no difference in the degree of cellularity, mitoses, or inflammatory infiltrate among the outcome groups. All of the tumors expressed bax, and none expressed c-myc. Two (8%) were immunoreactive for p53, one of which recurred and one of which underwent MT. bcl-2 expression was observed in 9 (37%) of the IMTs, with no difference among the three groups. Two IMTs were aneuploid, one of which underwent MT. Neither morphologic evaluation for cellularity, mitosis, or inflammatory infiltrate nor expression of bax or c-myc were useful for prediction of clinical outcome. The combination of atypia, ganglion-like cells, p53 expression and DNA ploidy analysis, however, might be useful to identify IMTs that might undergo MT or pursue a more aggressive clinical course with recurrences.
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            Inflammatory myofibroblastic tumor in children: clinical review with anaplastic lymphoma kinase, Epstein-Barr virus, and human herpesvirus 8 detection analysis.

            Inflammatory myofibroblastic tumor (IMT) is considered as an intermediate neoplasm that may present malignant features. Differential diagnosis with other tumor processes is sometimes difficult. Similar anaplastic lymphoma kinase (ALK) gene abnormalities as in anaplastic large cell lymphoma have been reported. Human herpesvirus 8 (HHV-8) DNA sequences have been described in adult pulmonary IMTs and Epstein-Barr virus (EBV) has been reported in splenic and hepatic IMTs, suggesting the importance of both viruses in IMT development. This article aims to evaluate ALK, EBV, and HHV-8 expression in children with IMT and to correlate our findings with clinical features. Sixteen children (range, 1-15 years) who had surgery for IMT between 1978 and 2003 were evaluated retrospectively. Formalin-fixed, paraffin-embedded archival tissues were stained for HHV-8 and ALK with immunohistochemistry. Epstein-Barr virus was detected by in situ hybridization (EBER probes). Tumors were located in the pulmonary lobe (n = 4), urinary tract (n = 4), mesentery or bowel (n = 4), hepatic lobe (n = 1), vena cava (n = 1), spinal cord (n = 1), and soft tissue (n = 1). Five children were treated with steroids and/or antibiotics before surgery, with no substantial result. IMT was excised totally in all but 2 cases. Four patients presented aggressive IMT with recurrence or metastasis requiring new surgery. ALK was positive in 3 (18.8%) cases and EBV in 1 pulmonary and 1 bladder tumor, all of them without recurrence or metastasis. None of the cases were positive for HHV-8. All patients are now disease-free with a mean follow-up of 4.2 years. Considering the present lack of efficient medical treatment, surgery should still be considered as the mainstay therapy in IMT, even in cases of recurrence or metastases. Larger multicentric studies would be necessary to understand the prognostic significance of ALK, EBV, and HHV-8 and their relationships with the origins of the tumor.
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              Primary inflammatory myofibroblastic tumor of the breast with rapid recurrence and metastasis: A case report.

              Primary inflammatory myofibroblastic tumor (IMT) of the breast is extremely rare; only 19 cases have been reported in the English literature. In the present study, we present a case of IMT in a 56-year-old female patient who was admitted to our hospital due to a mass found in her right breast. Mammogram and ultrasound revealed a well-circumscribed mass and surgery was performed. Histopathologically, the lesion was composed of spindle and inflammatory cells, including plasma cells and lymphocytes. Mitotic figures were not observed. Immunohistochemically, the tumor cells were positive for SM-actin, anaplastic lymphoma kinase (ALK) and vimentin and focal positive for desmin, but negative for NSE, S-100, CD117, CD34, NF, CD21, CD35 and CD68. Thus, we made a diagnosis of IMT and advised regular follow-up. However, the patient had local recurrence and metastasis to the left groin area 3, 7 and 10 months after the initial surgery. Notably, the histopathological characteristics of the recurrent and metastatic foci were similar to those of the initial specimen, but mitotic figures were clearly observed. Thus, we conclude that IMT shows occasionally malignant biological behavior although it is a neoplasm of intermediate biological potential that frequently recurs and rarely metastasizes. We advise that clinical physicians should regularly follow up patients after focal resection for IMT.
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                Author and article information

                Journal
                J Breast Cancer
                J Breast Cancer
                JBC
                Journal of Breast Cancer
                Korean Breast Cancer Society
                1738-6756
                2092-9900
                June 2015
                26 June 2015
                : 18
                : 2
                : 200-205
                Affiliations
                Department of Radiology, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, Jeonju, Korea.
                [1 ]Department of Pathology, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, Jeonju, Korea.
                [2 ]Department of Surgery, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, Jeonju, Korea.
                Author notes
                Correspondence to: Gong Yong Jin. Department of Radiology, Research Institute of Clinical Medicine of Chonbuk National University, Biomedical Research Institute of Chonbuk National University Hospital, Institute for Medical Sciences of Chonbuk National University Medical School, 20 Geonji-ro, Deokjin-gu, Jeonju 561-712, Korea. Tel: +82-63-250-2307, Fax: +82-63-272-0481, gyjin@ 123456chonbuk.ac.kr
                Article
                10.4048/jbc.2015.18.2.200
                4490271
                26155298
                e171db3c-0f94-4f89-bd3a-761d1fe39495
                © 2015 Korean Breast Cancer Society. All rights reserved.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 01 January 2015
                : 04 March 2015
                Categories
                Case Report

                Oncology & Radiotherapy
                breast neoplasms,granuloma,metastasis,plasma cells
                Oncology & Radiotherapy
                breast neoplasms, granuloma, metastasis, plasma cells

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