Cytopathologic features of epithelioid inflammatory myofibroblastic sarcoma with correlation of histopathology, immunohistochemistry, and molecular cytogenetic analysis

Lee, Jen‐Chieh; Wu, Jiann‐Ming; Liau, Jau–Yu; Huang, Hsuan‐Ying; Lo, Cheng-Yu; Jan, I-Shiow; Hornick, Jason L.; Qian, Xiaohua

doi:10.1002/cncy.21558

Cited by 22 publications

(10 citation statements)

References 32 publications

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“…IMTs usually present as a solid mass originating from the lung and abdominopelvic region and have a tendency to affect children and young adults of both genders [1, 2]. Once treated as reactive inflammatory lesions, IMTs are better known now as solid tumors with intermediate malignant potential.…”

Section: Introductionmentioning

confidence: 99%

“…Once treated as reactive inflammatory lesions, IMTs are better known now as solid tumors with intermediate malignant potential. Though most IMTs can be treated by en bloc resection, the recurrence rate remains high, ranging from <2 to 25% in pulmonary and extrapulmonary sites, respectively, and metastasis occurs in <5% of cases, [1, 2] indicating that these unique cases are worth intensive study.…”

Section: Introductionmentioning

confidence: 99%

“…RANBP2-ALK -associated EIMS has attracted the most attention for its unique pathological and clinical manifestations. RAN binding protein 2 (RANBP2) is a small GTP-binding protein of the RAS superfamily that is mainly located in the nuclear membrane [2]. Patients with IMT harboring RANBP2-ALK have been reported to suffer from rapid local recurrences and a higher death rate.…”

Section: Introductionmentioning

confidence: 99%

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Identification of EML4-ALK as an alternative fusion gene in epithelioid inflammatory myofibroblastic sarcoma

Jiang

Tong

Hou

et al. 2017

Orphanet J Rare Dis

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BackgroundKnown as solid tumors of intermediate malignant potential, most inflammatory myofibroblastic tumors (IMTs) are treatable as long as the tumor is en-bloc resected. However, in some cases, the tumors have recurred and grown rapidly after successful surgery. Some of these tumors were classified as an epithelioid inflammatory myofibroblastic sarcoma (EIMS). Most previously reported EIMSs have been caused by RANBP2-ALK fusion gene. We herein report an EIMS case caused by an EML4-ALK fusion gene.MethodsRNAseq was conducted to find out the new ALK fusion gene which could not be detected following previously reported RT-PCR methods for EIMS cases with RANBP2-ALK fusion gene. After that, RT-PCR was also conducted to further prove the newly found fusion gene. Immunohistochemistry (IHC) and fluorescence in situ hybridization (FISH) test were applied to find out the unique morphological characters compared with the previous reported EIMS cases.ResultsWe found an EIMS case who was suffering from a rapid recurrence after cytoreducyive surgery was done to relieve the exacerbating symptoms. The patient finally died for tumor lysis syndrome after the application of crizotinib. Distinctive ALK staining under the membrane and relatively weak ALK staining in the cytoplasm could also be observed. RNAseq and RT-PCR further revealed that the tumor harbored an EML4-ALK fusion gene.ConclusionIn conclusion, this is the first EIMS demonstrated to have been caused by the formation of an EML4-ALK fusion gene. This enriches the spectrum of EIMS and enlarges the horizon for the study of EIMS. The experience we shared in managing this kind of disease by discussing aspects of its success and failure could be of great value for surgeons and pathologists.Electronic supplementary materialThe online version of this article (doi:10.1186/s13023-017-0647-8) contains supplementary material, which is available to authorized users.

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Identification of EML4-ALK as an alternative fusion gene in epithelioid inflammatory myofibroblastic sarcoma

Jiang

Tong

Hou

et al. 2017

Orphanet J Rare Dis

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“…Intraoperative touch imprints are invaluable tools for assessing the cytological details especially for sites inaccessible for FNAC. In cases of intraoperative diagnosis of a malignant lesion, metastatic work up and additional treatment modalities can be devised . Thus, correlation between imprint cytological diagnosis and histology is of great importance.…”

Section: Discussionmentioning

confidence: 99%

Cytodiagnosis of inflammatory myofibroblastic tumor: A report of three cases in infants

Ghosh

Islam

Saha

et al. 2018

Diagnostic Cytopathology

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Inflammatory myofibroblastic tumor is a tumor of intermediate grade with a low rate of metastasis. The tumor often mimics malignancy. There is sparse literature available describing the cytological findings of this rare condition. It often presents in infancy and childhood as an intra-abdominal mass. Here, we describe the cytological findings of three cases of inflammatory myofibroblastic tumor in infants aged 10 months, 4 months, and 15 months, respectively. While the first two cases are fine needle aspiration cytology (FNAC) smears, the third case is that of an intraoperative imprint cytology. All the three smears showed low cellularity with small clusters of bland spindle cells, along with sprinkling of inflammatory cells, suggestive of an inflammatory myofibroblastic tumor. The diagnosis was later confirmed on histopathology and positive immunostaining for ALK.

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“…Older patients may present with epithelioid variants of myxofibrosarcoma and pleomorphic liposarcoma; the characteristic myxoid stroma and curvilinear vessels of myxofibrosarcoma or large lipoblasts of pleomorphic liposarcoma are helpful clues, if present. The epithelioid variant of inflammatory myofibroblastic tumor is an aggressive subtype that commonly presents as large intra‐abdominal tumors in young adult men and shows large atypical epithelioid cells, myxoid stroma, neutrophils, and delicate intervening vessels . ALK shows a nuclear membranous staining pattern secondary to ALK ‐ RANBP2 fusion; tumors also show CD30 and desmin positivity.…”

Section: Soft Tissue Tumors With Epithelioid Morphologymentioning

confidence: 99%

Application of ancillary studies in soft tissue cytology using a pattern‐based approach

Chebib

2018

Cancer Cytopathology

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The cytologic diagnosis of soft tissue neoplasms has undergone many advances due to the development of numerous useful immunohistochemical markers and molecular assays that target specific molecular alterations. Ancillary tests, when used in conjunction with clinical, radiologic, and cytomorphologic features, allow for more accurate classification and refinement of differential diagnoses of soft tissue neoplasms. Soft tissue tumors encompass a diverse group of tumors showing a wide range of cytomorphologies that can be broadly grouped as lipomatous, myxoid, spindled, pleomorphic, round cell, and epithelioid. This pattern-based classification enables the formation of appropriate differentials; however, entities that share these basic morphologic patterns may vary greatly in their clinical behavior. The breadth of soft tissue tumors that can be diagnosed on cytologic preparations continues to increase with identification of defining molecular alterations that can be detected by conventional means (eg, fluorescence in situ hybridization) or by immunohistochemical surrogates. Furthermore, the emergence of high-throughput molecular techniques (eg, next generation sequencing) has potential roles for additional discovery and diagnostic applications. This review highlights the diagnostic role of ancillary tests in the context of a pattern-based approach to soft tissue cytology. Cancer Cytopathol 2018;000:000-000. © 2018 American Cancer Society.

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Cytopathologic features of epithelioid inflammatory myofibroblastic sarcoma with correlation of histopathology, immunohistochemistry, and molecular cytogenetic analysis

Cited by 22 publications

References 32 publications

Identification of EML4-ALK as an alternative fusion gene in epithelioid inflammatory myofibroblastic sarcoma

Identification of EML4-ALK as an alternative fusion gene in epithelioid inflammatory myofibroblastic sarcoma

Cytodiagnosis of inflammatory myofibroblastic tumor: A report of three cases in infants

Application of ancillary studies in soft tissue cytology using a pattern‐based approach

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