Case Report


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

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1 Department of Diagnostic Pathology, National Hospital Organization, Fukuyama Medical Center, Fukuyama, Japan

2 Department of Otolaryngology-Head and Neck Surgery, National Hospital Organization, Fukuyama Medical Center, Fukuyama, Japan

3 Department of Diagnostic Pathology, Okayama University Hospital, Japan

4 Department of Pathology and Translational Research, Gifu University Graduate School of Medicine, Gifu, Japan

Address correspondence to:

Hiroshi Sonobe

Department of Diagnostic Pathology, National Hospital Organization, Fukuyama Medical Center, Fukuyama,

Japan

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Article ID: 100114Z10HS2022

doi: 10.5348/100114Z10HS2022CR

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How to cite this article

Sonobe H, Omote R, Fukushima K, Yanai H, Niwa R, Saigo C. A case of thyroid sclerosing mucoepidermoid carcinoma with eosinophilia indicates interleukin-5 production in a man with a history of Graves’ disease. J Case Rep Images Oncology 2022;8(2):37–44.

ABSTRACT


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.

Keywords: Interleukin-5 production, Graves’ disease, Male, Thyroid sclerosing mucoepidermoid carcinoma with eosinophilia

SUPPORTING INFORMATION


Acknowledgments

We thank all the staffs of the Department of Diagnostic Pathology, NHO Fukuyama Medical Center for various support for this case.

Author Contributions

Hiroshi Sonobe - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Rika Omote - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Kei Fukushima - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Hiroyuki Yanai - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Riko Niwa - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Chiemi Saigo - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published

Guaranter of Submission

The corresponding author is the guarantor of submission.

Source of Support

None

Consent Statement

Written informed consent was obtained from the patient for publication of this article.

Data Availability

All relevant data are within the paper and its Supporting Information files.

Conflict of Interest

Authors declare no conflict of interest.

Copyright

© 2022 Hiroshi Sonobe et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.