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Primary Thyroid Mucoepidermoid Carcinoma (MEC) Is Clinically, Prognostically, and Molecularly Different from Sclerosing MEC with Eosinophilia: A Multicenter and Integrated Study

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Abstract

Mucoepidermoid carcinoma (MEC) and sclerosing MEC with eosinophilia (SMECE) are rare primary thyroid carcinomas. In this study, we aimed to present our multicenter series of MEC and SMECE and integrated our data with published literature to further investigate the clinicopathological characteristics and prognoses of these tumors. We found 2 MECs and 4 SMECEs in our multicenter archives. We performed fluorescence in situ hybridization (FISH) to determine the MAML2 gene rearrangement. We screened for mutations in BRAF, TERT promoter, and RAS mutations using Sanger sequencing and digital polymerase chain reaction. Histopathologically, MECs and SMECEs were composed of two main cell types including epidermoid and mucin-secreting cells, arranged in cords, nests, and tubules. SMECEs were characterized by a densely sclerotic stroma with abundant eosinophils. We did not detect any MAML2 fusion in any of our cases. Two MEC cases harbored concomitant BRAF p.V600E and TERT C228T mutations. RAS mutations were absent in all cases. Concurrent foci of another thyroid malignancy were more commonly seen in MECs (p < 0.001), whereas SMECEs were associated with chronic lymphocytic thyroiditis (p < 0.001). MECs and SMECEs had equivalent recurrence-free survival (RFS) but MECs conferred significantly dismal disease-specific survival (DSS) as compared to SMECEs (p = 0.007). In conclusion, MECs and SMECEs not only shared some similarities but also demonstrated differences in clinicopathological characteristics, prognoses, and molecular profiles. SMECEs had a superior DSS in comparison to MECs, suggesting that they are low-grade cancers. This could help clinicians better evaluate patient outcomes and decide appropriate treatment plans.

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Data Availability

All data supporting this study are provided in full in the “Results” and “Supplementary” sections of this paper.

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Acknowledgements

We thank Dr. Terence Dunn (Department of Pathology, OUHSC) for assistance with the BRAF p.V600E testing.

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    1. Department of Pathology, University of Medicine and Pharmacy at Ho Chi Minh City, Ho Chi Minh City, 700-000, Vietnam

      Hieu Trong Le

    2. Department of Pathology, Chulalongkorn University, Krung Thep Maha Nakhon , Bangkok, 10300, Thailand

      Truong P. X. Nguyen

    3. Department of Diagnostic Pathology and Cytology, Kuma Hospital, Kobe, Hyogo, 650-0011, Japan

      Mitsuyoshi Hirokawa

    4. Department of Pathology, Ito Hospital, Shibuya, Tokyo, 150-8308, Japan

      Ryohei Katoh

    5. Department of Radiation Medical Sciences, Atomic Bomb Disease Institute, Nagasaki University, Nagasaki, 852-8523, Japan

      Norisato Mitsutake, Michiko Matsuse & Ayaka Sako

    6. Department of Pathology, University of Yamanashi, Yamanashi, 409-3821, Japan

      Tetsuo Kondo

    7. Department of Otolaryngology, Oklahoma University Health Sciences Center, Oklahoma City, OK, 73104, USA

      Nilesh Vasan

    8. Genetics Laboratory, Oklahoma University Health Sciences Center, Oklahoma City, OK, 73104, USA

      Young Mi Kim & Ying Liu

    9. Department of Pathology, Oklahoma University Health Sciences Center, Oklahoma City, OK, 73104, USA

      Lewis Hassell

    10. Department of Pathology, Cancer Genome Center and Thyroid Disease Center, Izumi City General Hospital, Izumi, Japan

      Kennichi Kakudo

    11. Department of Pathology, University of Iowa Hospitals and Clinics, Iowa City, IA, 52242, USA

      Huy Gia Vuong

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    HTL: data curation, formal analysis, investigation, methodology, writing original, and review. TPXN: data curation, formal analysis, investigation, methodology, writing original, and review. MH: data curation, formal analysis, investigation, methodology, and review. RK: data curation, formal analysis, investigation, methodology, and review. NM: data curation, formal analysis, investigation, methodology, and review. MM: data curation, formal analysis, investigation, methodology, and review. AS: data curation, formal analysis, investigation, methodology, and review. TK: data curation, formal analysis, investigation, methodology, and review. NV: data curation, formal analysis, investigation, methodology, and review. YMK: data curation, formal analysis, investigation, methodology, and review. YL: data curation, formal analysis, investigation, methodology, and review. LH: data curation, formal analysis, investigation, methodology, and review. KK: methodology and review. HGV: conceptualization, data curation, formal analysis, investigation, methodology, project administration, software, validation, writing original, review, and supervision. All authors have read and approved the manuscript.

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    Le, H.T., Nguyen, T.P.X., Hirokawa, M. et al. Primary Thyroid Mucoepidermoid Carcinoma (MEC) Is Clinically, Prognostically, and Molecularly Different from Sclerosing MEC with Eosinophilia: A Multicenter and Integrated Study. Endocr Pathol 34, 100–111 (2023). https://doi.org/10.1007/s12022-022-09741-1

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