Sclerosing Mucoepidermoid Carcinoma in the Parotid Gland With<i>CRTC1-MAML2</i>Fusion: A Case Report

Yabuki, Kei; Matsuyama, Atsuji; Shiba, Eisuke; Nagatani, Gunji; Hisaoka, Masanori

doi:10.1177/1066896917742721

Cited by 10 publications

(3 citation statements)

References 22 publications

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“…MEC cases were classified as follows: classical, clear cell, oncocytic, Warthin-like, spindle cell, sclerosing, central and others. 1,[12][13][14][15][16][17] In addition, high-grade transformation 18 and the recently described ciliated 19 and mucoacinar 20 variants were included in the variant list. In this process, central MEC was classified according to the site (intraosseous), and the other variants were classified according to the tumour histology.…”

Section: T U M O U R C L a S S I F I C A T I O Nmentioning

confidence: 99%

Salivary mucoepidermoid carcinoma: histological variants, grading systems, CRTC1/3‐MAML2 fusions, and clinicopathological features

et al. 2021

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Aims: To investigate the histological diversity of salivary mucoepidermoid carcinoma (MEC), its clinicopathological features, and its associations with CRTC1/ 3-MAML2 fusions. Methods and results: Salivary MEC cases (n = 177) were examined for CRTC1/3-MAML2 fusions, histological variants were classified, and tumours were graded according to four different grading systems. Adverse histological features considered to be unusual in MEC were also investigated. Of the 177 MEC cases, 110 were positive for CRTC1/3-MAML2 fusions. The classical variant was the most frequent in the fusion-positive case group, the fusion-negative case group, and the total case group. The clear/oncocytic variant was the second most frequent in the fusion-positive and total case groups. Oncocytic, Warthin-like and spindle variants were seen

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Section: T U M O U R C L a S S I F I C A T I O Nmentioning

confidence: 99%

Salivary mucoepidermoid carcinoma: histological variants, grading systems, CRTC1/3‐MAML2 fusions, and clinicopathological features

et al. 2021

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“…However, storiform fibrosis and obstructive phlebitis, which are specific to IgG4-related diseases, are not detected [30]. In another case, no significant IgG4 positive plasma cells were detected [13], and a sclerosing variant of MEC with the MALA2 translocation was reported [31]. Therefore, the mechanism of extensive fibrosis in SMECE is not fully understood.…”

Section: Discussionmentioning

confidence: 96%

A case of thyroid sclerosing mucoepidermoid carcinoma with eosinophilia indicates interleukin-5 production in a man with a history of Graves’ disease

Sonobe

Omote

Fukushima

et al. 2022

J Case Rep Images Oncology

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Thyroid sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE) is a rare tumor that typically affects women with Hashimoto’s thyroiditis. The present case was a man in his late 50s who was diagnosed with Graves’ disease at the age of 10 and was given antithyroid hormone for five years. The computed tomography scan revealed a nodular lesion in the right lobe, and the lesion was cytologically suspected as papillary carcinoma. No lymph node metastases or distant metastases were found. Before total thyroidectomy, high serum anti-thyroid peroxidase (TPO) and antithyroglobulin (TG) antibody titers, with no eosinophilia were detected. In a few small areas of the tumor center, small tumor cell foci with mild to moderate atypia, displaying mucous glandular cell and squamous cell differentiation, were found. The tumor was completely replaced by prominent sclerosing fibrosis, which was accompanied by tumor cell infiltration. The tumor had invaded the adjacent parenchyma and perithyroidal fatty tissue. In addition to lymphocytes and plasma cells, a large number of eosinophils were observed within the tumor. Immunohistochemically, tumor cells were strongly positive for p63, 34βE12, and TTF-1, but weakly for PAX8. Using fluorescence in situ hybridization (FISH), no MAML2 translocation was detected. Taken together with these findings, the present tumor was diagnosed as primary thyroid sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE). This case is the first to report thyroid SMECE associated with Graves’ disease. IL-5 immunostaining was performed to identify eosinophilia within the present tumor. As a result, the tumor cells were found to be positive for IL-5. The present tumor is also the first to indicate IL-5 production of SMECE.

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“…Though, a vast majority of MECs have classical appearance, morphological diversity occurs, albeit being less common. These variations are seen in the form of cellular and stromal features in MEC namely clear cell(6), oncocytic (7), ciliated(8), sclerosing (9), Warthin-like (10), spindle cell (11) and muco-acinar (12). Multiple cytological variations may occur within a single tumor such as the tumor may show areas of columnar, clear cell and/or oncocytic features (13).…”

Section: Introductionmentioning

confidence: 99%

Warthin-Like Mucoepidermoid Carcinoma: A Case Series Addressing the Need for Diagnostic Criteria Manuscript type: Case series

Agrawal,

Patil,

Mittal

et al. 2024

Preprint

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Warthin-like Mucoepidermoid carcinoma(WLMEC) is a newly described rare variant of Mucoepidermoid carcinoma(MEC). Rarity, coupled with potential misdiagnoses as the benign Warthin tumor necessitates careful examination and understanding of clinical and histological features. Lack of defining criteria in the current classification adds to the conundrum. Cases of WLMEC were retrieved from the archives of Department of Pathology from 2017-2022. Clinical and imaging data were obtained from the Electronic medical record system. A total of three cases, in two males and one female, aged 25, 49, and 64 years were diagnosed based on the proposed diagnostic criteria. A longstanding painless swelling in all cases, and lymphadenopathy in one case was seen at presentation. Imaging, in all cases, showed a hypodense mass with a mean diameter of 2 cm (range 1.1-2.9cm), involving the superficial lobe of parotid. Histomorphology, only superficially reminiscent of Warthin’s tumor at low power, was of a circumscribed tumour with disorganised cuboidal epithelium instead of oncocytic lining, with epithelial to lymphoid ratio varying from 0.4 to 0.9. Admixed areas with classic MEC morphology were seen (100% cases) comprising 5%, 15% and 25% of the tumour respectively. Atypical mitoses, lymphovascular invasion and perineurial invasion was not seen in any case. Immunohistochemistry for CK7 and P40 was diffusely positive in all. Mucicarmine stain highlighted interspersed mucous cells. As per MSKCC, AFIP and Brandwein systems, all these tumours were low grade, which corroborated with the prognosis as no recurrence or distant metastases was seen in a mean follow-up period of 12 months. Low power resemblance of WLMEC to Warthin tumor can lead to an erroneous diagnosis with prognostic implications, which may be obviated by using the proposed diagnostic criteria. Awareness of occurrence in younger age, presence of goblet cells, and areas with classic MEC morphology in a thoroughly sampled tumor might help avoid this pitfall.

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Sclerosing Mucoepidermoid Carcinoma in the Parotid Gland WithCRTC1-MAML2Fusion: A Case Report

Cited by 10 publications

References 22 publications

Salivary mucoepidermoid carcinoma: histological variants, grading systems, CRTC1/3‐MAML2 fusions, and clinicopathological features

Salivary mucoepidermoid carcinoma: histological variants, grading systems, CRTC1/3‐MAML2 fusions, and clinicopathological features

A case of thyroid sclerosing mucoepidermoid carcinoma with eosinophilia indicates interleukin-5 production in a man with a history of Graves’ disease

Warthin-Like Mucoepidermoid Carcinoma: A Case Series Addressing the Need for Diagnostic Criteria Manuscript type: Case series

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