Literature DB >> 34128136

Myoepithelial Carcinoma Ex-Pleomorphic Adenoma: A Rare Pathology Misdiagnosed as Pleomorphic Adenoma; With a Novel TERT Promoter Mutation and High PD-L1 Expression.

Christopher Cormier1, Shweta Agarwal2.   

Abstract

Myoepithelial carcinoma (MECA) is a rare salivary gland (SG) neoplasm (0.1-0.45% of all SG tumors) that often presents with bland cytomorphology and can be misclassified as cellular pleomorphic adenoma (PA) or myoepithelioma. This is particularly challenging in MECA ex-PA cases, especially if tumor shows minimal to no capsular invasion. We report a rare case of a 76-year-old female; history of left superficial parotidectomy with diagnosis (outside hospital) of cellular PA, who re-presented 9 months post surgery with enlarging left parotid mass, neck lymphadenopathy and facial nerve deficits. FNAB of parotid and neck lymph node revealed cellular aspirates with loosely cohesive clusters of myoepithelial cells with occasional chondromyxoid stroma. Prior resection slides were reviewed, and diagnosis of MECA ex-PA was made. Patient underwent left radical parotidectomy, selective neck dissection, with facial nerve sacrifice (due to extensive encasing by tumor). Histology showed a multinodular tumor with pushing borders, zonal arrangement comprising of a hypocellular, necrotic/myxoid center, and a peripheral rim of myoepithelial cells, confirmed by positive S100, and p63. Tumor extensively infiltrated peri parotid soft tissues with multiple foci of lymphovascular and perineural invasion; and metastatic neck lymph nodes. Next generation sequencing revealed a novel TERT promoter mutation (c.-124C > T), not usually described in SG neoplasms. Further, PD-L1 immunohistochemistry showed positive expression, making patient eligible for anti-PDL-1 immunotherapy. This case highlights importance of recognizing the subtle malignant features of MECA in distinguishing it from benign mimics like PA. In addition, presence of TERT mutation opens a new arena for future research to explore potential treatment targets.
© 2021. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Entities:  

Keywords:  Carcinoma ex-Pleomorphic adenoma; Myoepithelial; PD-L1; TERT

Mesh:

Substances:

Year:  2021        PMID: 34128136      PMCID: PMC9018951          DOI: 10.1007/s12105-021-01346-0

Source DB:  PubMed          Journal:  Head Neck Pathol        ISSN: 1936-055X


  27 in total

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Journal:  J Clin Invest       Date:  2019-10-01       Impact factor: 14.808

2.  Myoepithelial Carcinoma of the Salivary Glands: A Clinicopathologic Study of 29 Patients.

Authors:  Chenxing Wang; Zhuoyuan Zhang; Yaneng Ge; Zhe Liu; Jun Sun; Zhenjie Gao; Longjiang Li
Journal:  J Oral Maxillofac Surg       Date:  2015-04-01       Impact factor: 1.895

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Journal:  Bull World Health Organ       Date:  1981       Impact factor: 9.408

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Authors:  Sherif Said; John Campana
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5.  PLAG1 alteration in carcinoma ex pleomorphic adenoma: immunohistochemical and fluorescence in situ hybridization studies of 22 cases.

Authors:  Armita Bahrami; James D Dalton; Bangalore Shivakumar; Jeffrey F Krane
Journal:  Head Neck Pathol       Date:  2012-04-08

6.  Myoepithelial carcinoma arising in a pleomorphic adenoma of the parotid gland: report of a case with cytopathologic findings.

Authors:  Anil V Parwani; Giovanni Lujan; Syed Z Ali
Journal:  Acta Cytol       Date:  2006 Jan-Feb       Impact factor: 2.319

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Authors:  Takashi Mukaigawa; Ryuichi Hayashi; Kazuki Hashimoto; Toru Ugumori; Naohito Hato; Satoshi Fujii
Journal:  J Surg Oncol       Date:  2016-04-25       Impact factor: 3.454

9.  Myoepithelial tumors of salivary glands: a clinicopathologic, immunohistochemical, ultrastructural, and flow-cytometric study.

Authors:  L Alós; A Cardesa; J A Bombí; C Mallofré; A Cuchi; J Traserra
Journal:  Semin Diagn Pathol       Date:  1996-05       Impact factor: 3.464

10.  Cancer-associated TERT promoter mutations abrogate telomerase silencing.

Authors:  Kunitoshi Chiba; Joshua Z Johnson; Jacob M Vogan; Tina Wagner; John M Boyle; Dirk Hockemeyer
Journal:  Elife       Date:  2015-07-21       Impact factor: 8.140

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