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      Kidney inflammatory myofibroblastic tumor masquerading as metastatic malignancy: A case report and literature review

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          Abstract

          BACKGROUND

          Inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal tumor that is characterized by spindle cells differentiated from muscle fibroblasts and infiltration of various types of inflammatory cells. IMT can occur at any age and at any anatomic site. The most common location of IMT is the bladder in the genitourinary tract. Only scarce cases of kidney IMT have been reported in the literature.

          CASE SUMMARY

          A 77-year-old woman, with a history of bilateral renal calculus for 15 years, was admitted to the Department of Urology of our hospital complaining of recurrent painless gross hematuria for one month. The treatment with cephalosporin was ineffective. Computed tomography imaging showed a mixed density and slightly heterogeneously enhanced lesion in the middle pole of the left kidney and ipsilateral adrenal enlargement. The patient underwent surgical treatment by retroperitoneoscopic left radical nephrectomy plus adrenalectomy. A large number of typical spindle cells surrounded by plasma cells and lymphocytes were observed microscopically. Immunohistochemical analyses indicated that these spindle cells were positive for vimentin, cytokeratin (CK), Ki-67, CK7, CD34, and CD31 and were focally positive for CD10 and anaplastic lymphoma kinase (ALK-1). Thus, a diagnosis of IMT was made definitively. The patient recovered well after operation, and no recurrence or metastasis was noted during the 22-mo follow-up.

          CONCLUSION

          Since kidney IMT is very rare and lacks characteristic clinical manifestation, it is easily misdiagnosed as a malignant tumor before operation. Surgery remains the best choice for diagnosis and treatment, and such cases must be followed carefully because of the uncertain biological behavior of this tumor. This report suggests that renal calculus may be one of the causes of IMT, but further investigation is necessary to prove it.

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          Most cited references65

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          Inflammatory myofibroblastic tumors harbor multiple potentially actionable kinase fusions.

          Inflammatory myofibroblastic tumor (IMT) is a neoplasm that typically occurs in children. The genetic landscape of this tumor is incompletely understood and therapeutic options are limited. Although 50% of IMTs harbor anaplastic lymphoma kinase (ALK) rearrangements, no therapeutic targets have been identified in ALK-negative tumors. We report for the first time that IMTs harbor other actionable targets, including ROS1 and PDGFRβ fusions. We detail the case of an 8-year-old boy with treatment-refractory ALK-negative IMT. Molecular tumor profiling revealed a ROS1 fusion, and he had a dramatic response to the ROS1 inhibitor crizotinib. This case prompted assessment of a larger series of IMTs. Next-generation sequencing revealed that 85% of cases evaluated harbored kinase fusions involving ALK, ROS1, or PDGFRβ. Our study represents the most comprehensive genetic analysis of IMTs to date and also provides a rationale for routine molecular profiling of these tumors to detect therapeutically actionable kinase fusions.
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            Inflammatory Myofibroblastic Tumors: Current Update.

            Inflammatory myofibroblastic tumor (IMT) is a mesenchymal neoplasm of intermediate biological potential with a predilection for the lung and abdominopelvic region. IMT represents the neoplastic subset of the family of inflammatory pseudotumors, an umbrella term for spindle cell proliferations of uncertain histogenesis with a variable inflammatory component. IMTs show characteristic fasciitis-like, compact spindle cell and hypocellular fibrous histologic patterns and distinctive molecular features. Imaging findings reflect pathologic features and vary from an ill-defined, infiltrating lesion to a wellcircumscribed, soft tissue mass owing to variable inflammatory, stromal, and myofibroblastic components.
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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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                Author and article information

                Contributors
                Journal
                World J Clin Cases
                WJCC
                World Journal of Clinical Cases
                Baishideng Publishing Group Inc
                2307-8960
                26 December 2019
                26 December 2019
                : 7
                : 24
                : 4366-4376
                Affiliations
                Graduate School, Guangdong Medical University, Zhanjiang 524000, Guangdong Province, China
                Graduate School, Guangdong Medical University, Zhanjiang 524000, Guangdong Province, China
                Department of Urology, The Second Affiliated Hospital, Shenzhen University, Shenzhen 518100, Guangdong Province, China
                Department of Urology, The Second Affiliated Hospital, Shenzhen University, Shenzhen 518100, Guangdong Province, China. m13751317669@ 123456126.com
                Author notes

                Author contributions: Zhang GH was a major contributor in writing the manuscript; Guo XY searched the database and analyzed the data; Liang GZ collected and provided clinical data for the patient; Wang Q was responsible for editing the article; all authors read and approved the final manuscript.

                Supported by the Science and Technology Research and Development Foundation of Shenzhen, No. JCYJ20170307094039571.

                Corresponding author: Qing Wang, MD, PhD, Professor of Medicine, Department of Urology, The Second Affiliated Hospital, Shenzhen University, No. 118, Longjing Second Road, Baoan District, Shenzhen 518100, Guangdong Province, China. m13751317669@ 123456126.com

                Telephone: +86-1892655191

                Article
                jWJCC.v7.i24.pg4366
                10.12998/wjcc.v7.i24.4366
                6940336
                5dc26cb2-8842-47c6-93bf-1871f72f4e0c
                ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.

                This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial.

                History
                : 12 August 2019
                : 8 November 2019
                : 15 November 2019
                Categories
                Case Report

                inflammatory myofibroblastic tumor,kidney,diagnosis,renal calculus,case report

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