Case Report
Copyright ©The Author(s) 2018. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Oncol. Dec 20, 2018; 9(8): 200-207
Published online Dec 20, 2018. doi: 10.5306/wjco.v9.i8.200
Radiation-associated epithelial-myoepithelial carcinoma among five secondary malignancies: A case report and review of literature
Mohamed H Khattab, Alexander D Sherry, Carolyn G Ahlers, Austin N Kirschner
Mohamed H Khattab, Austin N Kirschner, Department of Radiation Oncology, Vanderbilt University Medical Center, Nashville, TN 37232, United States
Alexander D Sherry, Carolyn G Ahlers, Vanderbilt University School of Medicine, Nashville, TN 37232, United States
Author contributions: All authors contributed to the design and conceptualization of the report; all authors contributed to the collection and summarization of the patient’s clinical data; all authors drafted or revised the paper for important intellectual content; and all authors approved the final version of the paper.
Supported by the National Institutes of Health grant, No. 5K12CA090625-18 (to Austin N Kirschner) from the Vanderbilt Clinical Oncology Research Career Development Program; NCI/NIH Cancer Center Support Grant, No. 5P30 CA68485-19; and the Vanderbilt Mouse Metabolic Phenotyping Center Grant No. 2 U24 DK059637-16.
Informed consent statement: This study was exempt from IRB review per Vanderbilt University IRB Policy I.B.1. All identifiable patient information has been omitted, and the information contained in the manuscript is anonymized. The patient reported in this study gave verbal informed consent authorizing use and disclosure of his protected health information before his death.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author to: Austin N Kirschner, MD, PhD, Assistant Professor, Department of Radiation Oncology, Vanderbilt University Medical Center, 2220 Pierce Avenue, PRB-B1003, Nashville, TN 37232, United States. austin.kirschner@vumc.org
Telephone: +1-615-3222555 Fax: +1-615-3433075
Received: July 12, 2018
Peer-review started: July 12, 2018
First decision: October 8, 2018
Revised: November 7, 2018
Accepted: November 15, 2018
Article in press: November 15, 2018
Published online: December 20, 2018
Processing time: 162 Days and 10.2 Hours
Abstract
BACKGROUND

Epithelial-myoepithelial carcinoma (EMC) is a rare, low-grade, malignant tumor that constitutes less than one percent of all salivary gland tumors. To date, only one other case report has described radiation-associated EMC in the English language medical literature.

CASE SUMMARY

In this report, we describe the case of a 56-year-old male patient who presented with a neck mass diagnosed as EMC of the left submandibular gland approximately 30 years after mantle field radiation and chemotherapy for Hodgkin lymphoma. Treatment included resection, re-resection with nodal dissection, and adjuvant chemoradiotherapy. This patient was also diagnosed with 4 other secondary malignancies, including stage IV diffuse large B cell lymphoma in the abdomen with subsequent brain metastases, low-grade neuroendocrine carcinoma of the lung, Hurthle cell adenoma, and small B cell lymphoma before the patient expired. This case provides important information regarding the pathology, clinical sequelae, and management of a patient diagnosed with radiation-associated EMC amidst four concurrent malignancies.

CONCLUSION

Further investigation is needed on the efficacy of adjuvant radiotherapy in EMC, especially atypical EMC.

Keywords: Secondary malignancy; Radiation-associated malignancy; Epithelial-myoepithelial carcinoma; Cancer survivorship; Case report

Core tip: We describe the second known case of radiation-associated epithelial-myoepithelial carcinoma (EMC). This patient received chemotherapy and radiation therapy thirty years prior for Hodgkin lymphoma, and in addition to EMC was also diagnosed with four other malignancies in a span of five years. Because of worrisome histopathologic features atypical for EMC and pathologic stage of this patient, this patient was treated more aggressively with adjuvant chemoradiotherapy. This case provides important information regarding the pathology, clinical sequelae, and management of radiation-associated EMC. Further investigation is needed on the efficacy of adjuvant radiotherapy in EMC, especially atypical EMC.