Sclerosing mucoepidermoid carcinoma of salivary glands

Bacem Khalele Othman, Martina Bradova, Roderick H. W. Simpson, Jan Laco, Abbas Agaimy, Miguel Rito, Stephan Ihrler, Petr Steiner, Petr Grossmann, Veronika Hajkova, Gisele de Rezende, Montse Goma, Senada Koljenovic, Isabel Fonseca, Michal Michal, Ilmo Leivo, Alena Skalova*

*Corresponding author for this work

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Abstract

Sclerosing mucoepidermoid carcinoma (SMEC) of the salivary glands is a rare variant of low-grade mucoepidermoid carcinoma with scanty cellular atypia characterized by marked fibrosis/sclerosis and a rich inflammatory infiltrate. Herein, we report 25 unpublished cases of SMEC, two of them with prominent eosinophilia (2/25; 8%) and three with abundant IgG4-positive plasma cells (3/25; 12%). In our series of salivary SMEC, molecular analysis using fluorescence in situ hybridization (FISH) and/or next-generation sequencing (NGS) provided evidence of MAML2 gene rearrangement in 18 cases of the 21 analyzable cases tested (86%), while this gene locus was intact in 3 cases (14%). This study focuses on the diagnostic criteria of salivary SMEC given its challenge of abundant collagenous stroma, minimal residual neoplastic areas, and inconspicuous mucous cells. Follow-up data of our cases indicate that salivary SMECs have favorable outcomes. Molecular analysis for MAML2 gene rearrangement suggests that SMECs of salivary glands represent a rare variant of conventional low-grade MECs of salivary glands. In contrast, SMECs of the thyroid gland are genetically distinct from salivary-type thyroid MECs.
Original languageEnglish
Number of pages14
JournalVirchows Archiv
DOIs
Publication statusE-pub ahead of print - 14 Nov 2024

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