Sclerosing mucoepidermoid carcinoma with eosinophilia of the thyroid: more aggressive than previously reported

Quiroga‐Garza, Gabriela; Lee, Jeong H yeon; El‐Naggar, Adel K.; Black, Jennifer O.; Amrikachi, Mojgan; Zhai, Qihui; Ayala, Alberto G.; Ro, Jae Y.

doi:10.1016/j.humpath.2015.01.012

Cited by 36 publications

(25 citation statements)

References 26 publications

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“…A recent study has alluded to a potential for aggressive clinical behavior. 6 Additional key questions of interest include whether these tumors are biologically related to follicular derived thyroid carcinomas, or more to their salivary gland mucoepidermoid carcinoma counterparts, particularly with respect to CRCT1/3 - MAML2 translocation status. We present the clinicopathologic and molecular findings in nine cases of sclerosing mucoepidermoid carcinoma with eosinophilia and review the literature to expand our understanding of this rare entity.…”

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confidence: 99%

Thyroid sclerosing mucoepidermoid carcinoma with eosinophilia: a clinicopathologic and molecular analysis of a distinct entity

et al. 2017

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Sclerosing mucoepidermoid carcinoma with eosinophilia is a rare thyroid neoplasm of uncertain pathogenesis that resembles salivary gland mucoepidermoid carcinoma. This multi-institutional study characterizes the clinicopathologic and molecular features of this tumor by utilizing next-generation sequencing to assess common mutations and gene fusions involved in thyroid carcinogenesis as well as fluorescence in-situ hybridization for MAML2 translocations typical of salivary gland mucoepidermoid carcinoma. Nine cases (6 females and 3 males, mean age: 59 years, range 30–77 years) were identified. All cases were comprised of nests and strands of tumor cells with both squamous and mucinous differentiation embedded in a fibrohyaline stroma with an inflammatory infiltrate replete with eosinophils. All cases were p63 positive, thyroglobulin negative and showed variable expression of TTF-1. All nine cases were negative for MAML2 rearrangements. Five cases successfully tested by next-generation sequencing (ThyroSeq v.2 assay) were negative for mutations and translocations commonly involved in thyroid carcinogenesis. NTRK1 showed overexpression but no evidence of translocation. On follow-up, one patient died of persistent disease, whereas one of four remaining patients with available follow-up (mean: 7.3 years, range 4–11 years) demonstrated recurrence at 4 years. Thus, we show that sclerosing mucoepidermoid carcinoma with eosinophilia appears molecularly and morphologically distinct from follicular and C-cell-derived thyroid tumors as well as from salivary gland mucoepidermoid carcinoma. The overall and recurrence-free survival for these patients may be lower than for other well-differentiated thyroid cancers.

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confidence: 99%

Thyroid sclerosing mucoepidermoid carcinoma with eosinophilia: a clinicopathologic and molecular analysis of a distinct entity

et al. 2017

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“…Microscopically, thyroid SMECE is characterized by a combination of nested epidermoid carcinoma cells, intermediate carcinoma cells, and mucous-secreting carcinoma cells, fibrohyaline stroma, and prominent eosinophilic and lymphoplasmacytic infiltrates [10][11][12][13][14][15][16][17][18][19]. All of our cases showed these features.…”

Section: Discussionmentioning

confidence: 81%

“…[16]. The prognosis of this malignancy is favorable, although nodal metastases, extrathyroidal invasion, vascular invasion, and perineural invasion are common [12]. We encountered three patients with this rare sclerotic variant of MEC.…”

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confidence: 97%

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Thyroid sclerosing mucoepidermoid carcinoma with eosinophilia distinct from the salivary type

et al. 2018

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Abstract. We report three cases of thyroid sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE), which is an extremely rare variant of mucoepidermoid carcinoma (MEC). The aims of this report were to describe the clinicopathological findings, including results from immunohistochemical and fluorescence in situ hybridization analysis of thyroid SMECE, as well as to discuss the distinction between thyroid SMECE and its salivary counterpart. The cases included a 63-year-old female, a 44-year-old male, and a 66-year-old female, with all patients presenting with Hashimoto's thyroiditis. Nodal metastasis was not found in any of the three cases. Neither regional recurrences nor distant metastases were found in any patient during the follow-up, which was 20 years, 3 years, and 18 months, respectively. Histologically, tumors were composed of epidermoid carcinoma cells, intermediate type carcinoma cells, and goblet cell-type mucus-secreting carcinoma cells, with all tumors displaying a sclerotic stroma with eosinophilic and lymphocytic infiltration. The formation of eosinophilic abscess in the tumor nests that might be a novel characteristic finding of SMECE was observed. Immunohistochemically, the carcinoma cells were positive for cytokeratin 34βE12, TTF-1, and PAX8, but negative for thyroglobulin. In two cases, increased IgG4-positive plasma cells were observed. Mastermind-like transcriptional coactivator 2 (MAML2), according to fluorescence in situ hybridization, was intact in all cases. In conclusion, thyroid SMECE has favorable outcomes and seems to be genetically different from salivary MEC. This is the first report to describe the presence of increased IgG4-positive plasma cells in the stroma of SMECE. [16]. The prognosis of this malignancy is favorable, although nodal metastases, extrathyroidal invasion, vascular invasion, and perineural invasion are common [12]. We encountered three patients with this rare sclerotic variant of MEC. The aims of this report were to describe the clinicopathological findings, including results from immunohistochemical and fluorescence in situ hybridization (FISH) analysis of thyroid SMECE, as well as to discuss the distinction between thyroid SMECE and its salivary counterpart.

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“…7 However a recent study by Quiroga-Garza G et al suggests that SMECE of thyroid is more aggressive than previously thought. 3 They found distant metastases in 50% of their patients with SMECE of the thyroid. Further, a literature review in 2004 by Shehadeh et al found 4 out of 23 patients with SMECE of thyroid also had distant metastatic disease.…”

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confidence: 98%

An aggressive case of sclerosing mucoepidermoid carcinoma with eosinophilia of thyroid: a case report

Eason

Allison

2017

Int Surg J

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Sclerosing mucoepidermoid thyroid carcinoma with eosinophilia (SMECE) of the thyroid is a rare disease with approximately 50 cases reported worldwide. We describe SMECE of the thyroid affecting a 44 year old female and the first reported case of SMECE of thyroid in New Zealand. This case is of interest as the disease course was particularly aggressive with uncommon immunohistology. Our patient presented with a 4 month history of a painless swelling in her right upper neck. CT revealed a diffusely enlarged thyroid with infiltration of the trachea. A total thyroidectomy, tracheal resection and bilateral neck dissection was performed. Histology showed nests and cords of moderately sized epithelioid cells with prominent nucleoli, with focal glands and tubules. Immunohistology was performed. CK14 and epithelial membrane antigen staining was focally positive. The patient went on to receive chemo-radiotherapy. A CT scan later revealed metastasis to her lungs. She died 7 months from her diagnosis. Our case provides an example of aggressive SMECE of thyroid.

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Sclerosing mucoepidermoid carcinoma with eosinophilia of the thyroid: more aggressive than previously reported

Cited by 36 publications

References 26 publications

Thyroid sclerosing mucoepidermoid carcinoma with eosinophilia: a clinicopathologic and molecular analysis of a distinct entity

Thyroid sclerosing mucoepidermoid carcinoma with eosinophilia: a clinicopathologic and molecular analysis of a distinct entity

Thyroid sclerosing mucoepidermoid carcinoma with eosinophilia distinct from the salivary type

An aggressive case of sclerosing mucoepidermoid carcinoma with eosinophilia of thyroid: a case report

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