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Pediatric Warthin-like Mucoepidermoid Carcinoma: Report of Two Cases with One Persistent/Recurrent as Conventional Mucoepidermoid Carcinoma

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Abstract

Mucoepidermoid carcinoma (MEC) is the most common primary salivary gland malignancy. While salivary gland neoplasia is rare in children, MEC is much more likely to occur in the pediatric population than Warthin tumor, a common benign salivary gland neoplasm associated with smoking and older age. The recently-reported Warthin-like variant of MEC bears a striking histologic resemblance to Warthin tumor, representing a potential diagnostic pitfall. Therefore, low-power observation of Warthin-like features in pediatric salivary gland tumors should prompt careful diagnostic consideration of Warthin-like MEC. Two cases of Warthin-like MEC in the parotid glands of teenaged patients were identified in the archives of the Department of Pathology at Children’s Medical Center in Dallas, Texas. Surgical material for each case was reviewed and both diagnoses were verified. Fluorescence in situ hybridization (FISH) for CRTC1–MAML2 fusion was performed in both cases. Histologically, neither tumor exhibited the classic bilayer of oncocytic epithelial cells characteristic of Warthin tumor. Instead, the neoplastic epithelial cells exhibited architectural and cytologic atypia, with mucous cells interspersed. CRTC1–MAML2 gene fusions were identified via FISH and confirmed the diagnosis of MEC in both cases. Of note is that the second patient’s tumor recurred with features of conventional MEC, indicating the potential for Warthin-like MEC to undergo this morphologic change. The present cases illustrate that Warthin-like MEC, like conventional MEC, may occur in the pediatric population. Pediatric and head and neck pathologists must be aware of this variant’s existence and diagnostic criteria to avoid misdiagnosis as benign Warthin tumor.

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The authors received no financial support for the research, authorship, and publication of this article.

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Author notes

  1. Elena V. Daoud and Anne C. McLean-Holden have contributed equally.

Authors and Affiliations

  1. Department of Pathology, University of Texas Southwestern Medical Center, 6201 Harry Hines Blvd, Dallas, TX, 75390, USA

    Elena V. Daoud, Anne C. McLean-Holden, Charles F. Timmons & Justin A. Bishop

  2. Department of Radiology, University of Texas Southwestern Medical Center, Dallas, TX, USA

    Cory M. Pfeifer

  3. ProPath, Dallas, TX, USA

    Bahram R. Oliai

  4. Department of Pediatric Pathology, Children’s Medical Center, Dallas, TX, USA

    Charles F. Timmons

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  1. Elena V. Daoud
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  2. Anne C. McLean-Holden
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Correspondence to Justin A. Bishop.

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Daoud, E.V., McLean-Holden, A.C., Pfeifer, C.M. et al. Pediatric Warthin-like Mucoepidermoid Carcinoma: Report of Two Cases with One Persistent/Recurrent as Conventional Mucoepidermoid Carcinoma. Head and Neck Pathol 14, 923–928 (2020). https://doi.org/10.1007/s12105-020-01156-w

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