Myoepithelioma‐like tumor of the vulvar region showing infiltrative growth and harboring only a few estrogen receptor‐positive cells: A case report

Kojima, Yui; Tanabe, Mikiko; Kato, Ikuo; Motoi, Toru; Kimura, M.; Sawazumi, Tomoe; Tanaka, Reiko; Chiba, Sawako; Otani, Masako; Inayama, Yoshiaki

doi:10.1111/pin.12765

Cited by 9 publications

(13 citation statements)

References 14 publications

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“…In contrast, INI1 expression was retained in all cases of MESTBs in our study. Moreover, EWSR1 rearrangement was not detected in the MELTVR cases consistent with previous reports 10,11,13 . Therefore, the combination of these findings will facilitate distinguishing MESTBs from MELTVRs.…”

Section: Discussionsupporting

confidence: 88%

“…We selected 13 cases of MESTB and one case of MELTVR (case 15) from the clinicopathological archives at the Department of Surgical Pathology, Sapporo Medical University Hospital, Japan. We also included one MELTVR case (case 14) that has previously been reported 13 . We reviewed hematoxylin and eosin‐stained slides for histological confirmation of MESTB and MELTVR.…”

Section: Methodsmentioning

confidence: 99%

“…Recently, studies have reported a unique tumor of the vulvar region designated myoepithelioma‐like tumors of the vulvar region (MELTVR) 10–13 . MELTVRs occur in the vulvar region including the labium majora, mons pubis and groin in women.…”

Section: Introductionmentioning

confidence: 99%

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Myoepithelioma of soft tissue and bone, and myoepithelioma‐like tumors of the vulvar region: Clinicopathological study of 15 cases by PLAG1 immunohistochemistry

Segawa

Sugita

Aoyama

et al. 2020

Pathology International

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We demonstrated the clinicopathological findings of 13 myoepitheliomas of soft tissue and bone (MESTBs) and two myoepithelioma‐like tumors of the vulvar region (MELTVRs), focusing on the association between nuclear atypia and clinical course, and the utility of immunohistochemistry (IHC) of pleomorphic adenoma gene 1 (PLAG1) for the pathological diagnosis of these tumors. Of the 13 MESTBs, eight, one and four cases exhibited mild, moderate and severe nuclear atypia, respectively. Two cases with venous invasion showed severe nuclear atypia and both died of advanced disease. Two MELTVR cases showed moderate nuclear atypia and had no evidence of disease after surgery. On IHC, 12 of 13 (92.3%) MESTBs showed PLAG1 immunoreactivity and none of the MELTVRs expressed PLAG1. In addition, MELTVRs showed loss of INI1 expression. In contrast, all MESTBs retained INI1 expression. Fluorescence in situ hybridization detected EWSR1, FUS and PLAG1 rearrangement in 5 (38.5%), 0 (0%) and 2 (15.4%) of the 13 MESTBs, respectively. No EWSR1, FUS and PLAG1 rearrangement were observed in the METLVRs. In conclusion, MESTBs with both severe nuclear atypia and venous invasion would be indicative of malignant potential. PLAG1 might be a useful IHC marker in MESTB diagnosis.

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Section: Discussionsupporting

confidence: 88%

Section: Methodsmentioning

confidence: 99%

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Myoepithelioma of soft tissue and bone, and myoepithelioma‐like tumors of the vulvar region: Clinicopathological study of 15 cases by PLAG1 immunohistochemistry

Segawa

Sugita

Aoyama

et al. 2020

Pathology International

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“…MELTVR is a rare neoplasm. Up to present, eleven cases of MELTVR have been reported in the literature [1][2][3]. The tumor is not classified according to the 4th edition of WHO classification of Soft Tissue and Bone Tumors [4].…”

Section: Discussionmentioning

confidence: 99%

Myoepithelioma-like tumor of the vulvar region: a case report in China and review of the literature

Gao

2020

Diagn Pathol

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Background: Myoepithelioma-like tumor of the vulvar region (MELTVR) is a recently described mesenchymal neoplasm which typically arising in vulvar regions of adult women. Case presentation: Here we report a case of a 65-year-old woman who presented with a 6-year history of subcutaneous mass in the vulvar region. The mass had recently increased in size continuously. Histologically, the tumor cells had an epithelioid to spindled shape. Epithelioid tumor cells proliferated singly or in a loosely cohesive manner with myxoid areas, while spindled tumor cells grew in diffuse sheets or storiform arrangements mainly in nonmyxoid areas. Immunohistochemically, the tumor cells were positive for vimentin, epithelial membrane antigen, calponin, and were partially mild to moderate positive for estrogen receptor, but completely negative for S100 protein, glial fibrillary acidic protein, CD34, desmin, SMA and cytokeratin. INI1/SMARCB1 expression was deficient. EWSR1 and FUS genes were intact tested by fluorescence in situ hybridization analysis. Based on these findings, we diagnose this case as MELTVR. The patient remained relapse-free after the lesion was widely excised during 8 months follow-up. Conclusions: This disease should be included in the differential diagnostic list of vulvar tumors with epithelioid to spindled morphology. Recognition of its histopathological features and immunohistochemical reactivity will help to understand the tumor better.

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“…Occasional cases were reported to show minimal micronodular extracapsular infiltration, 1 while one case demonstrated extensive infiltrative growth and positive surgical margins. 5 Local, non-destructive recurrence has also been reported in incompletely excised lesions, with a case of late recurrence after 12 years. 1 The behaviour of MELTVR appears to be indolent, as no cases of metastatic disease have been reported, which is unusual as high grade nuclear atypia, vascular invasion, necrosis and increased mitotic activity have been observed in some cases.…”

Section: Discussionmentioning

confidence: 99%

Myoepithelioma‐like tumour of the vulval region: A case report

et al. 2021

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Myoepithelioma-like tumours of the vulvar region (MELTVR) are a newly described group of spindle cell neoplasm. Morphologically, they consist of epithelioid to spindled cells in a myxoid to collagenous stroma and can resemble epithelioid sarcomas, myoepithelial carcinomas or extraskeletal myxoid chondrosarcomas. However, they have a distinct pattern of immunohistochemical staining characterised by positivity for EMA, ER, PR and negativity for cytokeratin, GFAP and S100. Nuclear staining for INI-1 is lost. In addition, they lack the characteristic gene arrangements of these other lesions. MELTVR can recur locally when incompletely excised, however, do not appear to metastasize. We report a case of a MELTVR in a 49-year-old woman arising in the right mons pubis.

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Myoepithelioma‐like tumor of the vulvar region showing infiltrative growth and harboring only a few estrogen receptor‐positive cells: A case report

Cited by 9 publications

References 14 publications

Myoepithelioma of soft tissue and bone, and myoepithelioma‐like tumors of the vulvar region: Clinicopathological study of 15 cases by PLAG1 immunohistochemistry

Myoepithelioma of soft tissue and bone, and myoepithelioma‐like tumors of the vulvar region: Clinicopathological study of 15 cases by PLAG1 immunohistochemistry

Myoepithelioma-like tumor of the vulvar region: a case report in China and review of the literature

Myoepithelioma‐like tumour of the vulval region: A case report

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