Sclerosing Mucoepidermoid Carcinoma With Eosinophilia Research Articles

Cytopathology of primary sclerosing mucoepidermoid carcinoma with eosinophilia of the thyroid: a multi-institutional case series and review of literature

Sclerosing Mucoepidermoid Carcinoma with Eosinophilia (SMECE) of the thyroid is an extremely rare tumor that exhibits unique histologic characteristics and is nearly always associated with lymphocytic thyroiditis (LT). However, the cytomorphologic and clinicopathologic characteristics of SMECE have only been described in rare case reports. Authors' institution laboratory information systems were searched for records of SMECE between 2012 and 2023. Literature review was performed using keywords "Sclerosing mucoepidermoid carcinoma with eosinophilia", "thyroid", and "cytopathology" to search through institution electronic library databases for relevant articles. A total of 19 cases were identified, 3 unpublished in the authors' archives and 16 in the literature which had fine needle aspiration (FNA) material or cytologic features available for review, and were comprised of 3 males and 16 females. The common cytomorphologic characteristics of SMECE included fragments or loose clusters of intermediate-type epidermoid cells in a background of prominent LT and eosinophils. Overt keratinization, mucinous cells, and extracellular mucin were not commonly encountered, resulting in diagnostic challenges, especially if eosinophils associated with epithelial cell clusters were rare. The cases were reported as "Nondiagnostic" (1 case), "Atypia of Undetermined Significance" (4 cases), "Suspicious for Malignancy" (3 case), or "Malignant" (11 cases). The clinical course of SMECE of the thyroid varied and distinct cytomorphologic characteristics in a subset of patients who experienced aggressive disease raises the possibility of different prognostic grades. Cases with keratinized squamous cells and necrosis mimic anaplastic (undifferentiated) thyroid carcinoma, but the clinical history and radiologic findings can be helpful to exclude this diagnosis.

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

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.

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

Sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE) of the thyroid is a rare traditionally "low-grade" tumor that predominantly occurs in women. Approximately 50 cases have been reported in the literature. It arises in a background of Hashimoto thyroiditis and is characterized by nests of epidermoid and mucin-secreting cells located within an eosinophil-rich sclerotic stroma. Herein, we outline the clinicopathological and immunohistochemical characteristics of 6 cases of thyroid SMECE. All tumors were detected in women (age, 36-89 years; average, 59 years), and all patients underwent total thyroidectomies. Clinicopathological findings included extensive tumor invasion into the adjacent soft tissues, trachea, pharynx, and esophagus. Of 6 specimens, 5 had positive surgical margins. Cervical lymph node metastases were seen in 4, and distant metastases were in 3 patients. Immunohistochemically, all tumors were positive for CK19, galectin 3, and p63 and negative for calcitonin, calponin, S-100, and smooth muscle actin. Interestingly, 2 tumors also showed faint focal staining for thyroglobulin, and 2 others had focal positivity for thyroid transcription factor 1. Together, galectin 3 and CK19 expression supported the malignancy of these lesions, and p63 expression raised the possibility that these tumors originated from the ultimobranchial body. In summary, SMECE tumors in our series exhibit a clear female predominance with aggressive behavior and appear to arise from pluripotent solid cell nests. A correct diagnosis is crucial to providing SMECE patients with the appropriate treatment options, and we recommend a closer follow-up schedule than previously considered.

Eosinophilic Replacement Infiltrates in Cystic Hashimoto’s Thyroiditis: a Potential Diagnostic Pitfall

A 50-year-old woman underwent a fine-needle aspiration biopsy for progressive enlargement of the left thyroid lobe which was cystic and solid on ultrasound exam. The smears contained innumerable eosinophilic leukocytes along with lymphocytes, Hurthle cells, cells from a papillary thyroid carcinoma (PTC), and atypical glandular and squamous cells. The cytologic interpretation was Hashimoto's thyroiditis (HT), suspicious for epithelial neoplasm. The associated diagnostic comment stated concern for a sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE) arising in a PTC. Thyroidectomy demonstrated a PTC, HT with multiple lymphoepithelial cysts, and extensive multifocal infiltrates of eosinophils, generally confined to the cyst walls. As the cytologic findings mimicked a SMECE, we report these specimens as a most unusual diagnostic pitfall.

Sclerosing mucoepidermoid carcinoma with eosinophilia of the salivary gland: Case report and review of the literature

Sclerosing mucoepidermoid carcinoma with eosinophilia (SMCE) of the salivary gland is a rare variant of mucoepidermoid carcinoma. We report a case of SMCE in the right submandibular gland of a 79-year-old man. Fine needle aspiration cytology revealed cohesive clusters of atypical squamous epithelial cells admixed with cells containing intracytoplasmic mucin and eosinophils. Histologically, the tumor was composed of epithelial nests with keratinizing cells occasionally present at the center, as well as peripherally located atypical basaloid cells, and some mucin-containing cells embedded in a fibrosclerotic stroma, which were accompanied by a prominent lymphoplasmacytic and eosinophilic infiltrate. Inflammatory infiltrate and stromal fibrosclerosis also were seen in the non-neoplastic salivary gland tissue adjacent to the tumor. Immunohistochemically, many plasma cells were IgG4-positive. The postoperative serum IgG4 level was elevated. Our reverse transcription-polymerase chain reaction using formalin-fixed, paraffin-embedded tumor tissue failed to detect any fusion-gene transcripts which are specifically identified in ordinary mucoepidermoid carcinoma. The findings of the present case suggest that this rare type of salivary gland carcinoma may be associated with a chronic inflammatory condition such as IgG4-related sclerosing disease. Only 23 cases of sclerosing mucoepidermoid carcinoma with or without eosinophilic infiltratie have been reported to date in such an anatomical location.

Sclerosing mucoepidermoid carcinoma with eosinophilia of the thyroid

Sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE) is a recently described carcinoma of the thyroid gland associated with Hashimoto's thyroiditis and considered to have a relatively indolent clinical course. We present a 65-year-old female, clinically euthyroid, who presented with a thyroid swelling since last 8 months along with right-sided cervical lymphadenopathy. Fine needle aspiration cytology suggested poorly differentiated carcinoma along with the involvement of the lymphnodes while histopathological examination showed features of SMECE with metastatic deposits in the right-sided cervical lymphnodes.

Primary Mucoepidermoid Carcinoma and Sclerosing Mucoepidermoid Carcinoma with Eosinophilia of the Thyroid Gland: A Report of Nine Cases

Mucoepidermoid carcinoma is a rare primary thyroid tumor with indolent biologic potential. Two types of tumors have been described under this category: mucoepidermoid carcinoma (MEC) and sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE). The MEC shows both squamous and glandular differentiation in a background of a noninflamed gland, whereas SMECE is characterized by extensive sclerosis, squamous and glandular differentiation, a concomitant inflammatory infiltrate rich in eosinophils, and a background of lymphocytic thyroiditis.We present nine cases of these entities: five MEC and four SMECE. All tumors occurred in women (age 27 to 73 years). Five tumors showed extrathyroidal invasion and multiple lymph node metastases. One case of MEC showed a concomitant tall cell variant of papillary carcinoma with vascular invasion, and two cases showed intimately associated areas of usual papillary carcinoma. One of the latter cases also showed areas of transformation to anaplastic carcinoma. In all cases of SMECE and in only one case of MEC, the uninvolved thyroid tissue showed lymphocytic thyroiditis. Follow-up information was available in four of the nine cases (3 months to 7 years). Two patients with SMECE are alive with no evidence of disease. One patient with MEC and tall cell variant of papillary carcinoma died of disease after 3 months, and the patient with anaplastic carcinoma died after 5 months with lung metastasis.Both MEC and SMECE were positive for cytokeratin and negative for calcitonin. All cases of MEC were positive for thyroglobulin, whereas all cases of SMECE were negative. The immunohistochemical findings suggest that both MEC and SMECE have different histogenesis.

Sclerosing mucoepidermoid carcinoma with eosinophilia of the thyroid: Report of two patients, one with distant metastasis, and review of the literature

Sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE) is a recently recognized malignant neoplasm of the thyroid gland. Two additional cases of this condition which occurred in a 70-year-old woman and a 69-year-old woman are presented. The case of the 70-year-old woman (patient 1) is the first report of distant metastasis, besides lymph node metastasis, for this type of tumor. The patient initially presented with a thyroid mass, and the thyroid gland with surrounding cervical lymph nodes was removed. Because of focal keratin “pearl” formation, the tumor was misinterpreted as a metastatic squamous cell carcinoma to the thyroid. Approximately 4 years later, the patient developed a left supraclavicular mass and lung densities. A pathological fracture of the right humeral head followed, and the left supraclavicular mass recurred along with newly developed subcutaneous nodules on the chest wall and arm. Open lung and bone biopsies revealed metastatic SMECE, which was morphologically identical to that of the thyroid mass. The 69-year-old woman (patient 2) had, in 1983, undergone thyroidectomy with left radical neck dissection; this had been diagnosed as follicular carcinoma of the thyroid with lymph node involvement. After multiple isolated lymph nodes metastases, the patient developed locally extensive, recurrent tumor that showed microscopic features of SMECE. Review of the previous thyroid tumor and lymph nodes revealed the same type of histology. To our knowledge, only a single report containing eight cases of this distinctive carcinoma of the thyroid has been published. Herein we describe characteristic morphological features of two additional cases of this rare malignancy, one with distant metastasis, and we review the related literature.