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      Diagnostic Challenges in a Rare Submandibular Epidermoid Cyst Transforming into Squamous Cell Carcinoma - A Case Report

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          Abstract

          Rationale:

          Submandibular gland epidermoid cyst is extremely rare and malignant transformation into squamous cell carcinoma (SCC) is even rarer. No such case report is published.

          Patient Concerns:

          A 49-year-old female reported with a large, slowly growing left-sided neck swelling present for two years. Her primary concern was large swelling.

          Diagnosis:

          Clinical finding, cytology and imaging were suggestive of ‘Epidermoid Cyst of Submandibular Gland’. Intraoperative frozen section and post-operative definitive histopathological examination (HPE) revealed malignant transformation into SCC.

          Treatment:

          In toto surgical excision was performed. Once the frozen section HPE confirmed transformation into SCC, clearance of 4–6 mm margin and modified supraomohyoid neck dissection was completed.

          Outcome:

          No sign of recurrence on 1-year postoperative follow-up.

          Take-Away Lesson:

          This case is unique of its kind without any previous literature report. In case of large and rapidly growing submandibular gland lesion, possibility of malignant transformation should not be overruled.

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          Most cited references10

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          Epidermoid cyst of submandibular region

          Jananee Janarthanam, Sathishkumar Mahadevan (2012)
          Epidermoid cysts are rare, slow-growing, benign, developmental cysts that are derived from abnormally situated ectodermal tissue. Epidermoid cysts may grow anywhere on the body and about 7% of them are located in the head and neck. These cyst arise from traumatic implantation of epithelium or entrapment of epithelial remnants during embryonic fusion. Histopathologically, they are lined by stratified squamous epithelium and ,lumen without any skin appendages. Here we present a case of epidermod cyst occurring in sub-mandibular region.
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            Incidental Squamous Cell Carcinoma in an Epidermal Inclusion Cyst: A Case Report and Review of the Literature

            Ethan Frank, David Macías, Brian Hondorp … (2018)
            Epidermal inclusion cysts are common lesions that rarely develop into squamous cell carcinoma (SCC). Neoplastic change in these cysts can be associated with prominent symptoms such as pain, rapid growth, or ulceration. This study describes the case of a 64-year-old woman with a 4-year history of a largely asymptomatic neck mass, which after routine excision was found to be an epidermal inclusion cyst harboring well-differentiated SCC. The diagnosis was made incidentally after routine cyst bisection and hematoxylin and eosin staining. Given the potential for variable presentation and low cost of hematoxylin and eosin analysis, we recommend a low threshold for a comprehensive pathological search for malignancy in excised cysts when appropriate.
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              A Rare Transformation of Epidermoid Cyst into Squamous Cell Carcinoma: A Case Report with Literature Review

              Adel A. Faltaous, Emilia Leigh, Peter Ray … (2019)
              Patient: Male, 77 Final Diagnosis: Squamous cell carcinoma Symptoms: Epidermoid cyst Medication: — Clinical Procedure: Cyst excision Specialty: Plastic Surgery Objective: Rare co-existance of disease or pathology Background: Epidermoid cysts are the most common benign skin lesions that can occur anywhere on the body, and frequently occur on the face, scalp, neck, and trunk. Typically, patients are asymptomatic, and the lesions present as firm skin-colored nodules filled with keratinous or sebaceous materials that are formed by cystic expansion of the epidermal epithelium. Malignant transformation is rare in epidermoid cysts. Case Report: This is a case report of a 77-year-old male Caucasian patient who presented with an epidermoid cyst on the left parietal area. Due to high suspicion, the cyst was surgically excised and sent to pathology which resulted in discovery of malignant squamous cell carcinoma with unclear margins. Aimed at obtaining clear margins, a wide excision was performed, which revealed clean margins and absence of residual cancerous cells. The patient recovered well without major complications. Conclusions: Although the prevalence of epidermoid cysts is high, malignant transformation of epidermoid cysts into squamous cell carcinoma is rare. Due to its scarcity, the exact pathophysiology of malignant transformation is still poorly understood. The differential diagnosis should include the possibility of malignancy for highly suspicious lesions, and proper management guidelines should be established. Surgical resection should be the treatment of choice, and once removed, all surgically excised cysts should undergo pathologic evaluation. As clinicians, it is crucial to stay vigilant and have a low threshold for excision and thorough histological examination of specimens to allow early diagnosis and interventions which can significantly improve patient outcomes. Through the suggested guidelines, we hope to aid in better management and intervention in case of malignant transformation of epidermoid cysts.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Ann Maxillofac Surg
                Journal ID (iso-abbrev): Ann Maxillofac Surg
                Journal ID (publisher-id): AMS
                Journal ID (publisher-id): Ann Maxillofac Surg
                Title: Annals of Maxillofacial Surgery
                Publisher: Wolters Kluwer - Medknow (India )
                ISSN (Print): 2231-0746
                ISSN (Electronic): 2249-3816
                Publication date (Print): Jan-Jun 2023
                Publication date (Electronic): 27 March 2023
                Volume: 13
                Issue: 1
                Pages: 98-100
                Affiliations
                [1]Department of Oral and Maxillofacial Surgery, Army Dental Corps, New Delhi, India
                Author notes
                Address for correspondence: Dr. Ikbal Hossain, Flat No: C-4B, ANANDA Apartment, 8 Mall Road, Dumdum, Kolkata - 700 080, West Bengal, India. E-mail: piqlu1988@ 123456gmail.com
                Article
                Publisher ID: AMS-13-98
                DOI: 10.4103/ams.ams_167_22
                PMC ID: 10499289
                PubMed ID: 37711521
                SO-VID: 4622c298-6ce3-427e-aa7a-6e6e0cef50c0
                Copyright © Copyright: © 2023 Annals of Maxillofacial Surgery
                License:

                This is an open access journal, and articles are distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as appropriate credit is given and the new creations are licensed under the identical terms.

                History
                Date received : 04 September 2022
                Date revision received : 29 November 2022
                Date accepted : 13 December 2022
                Categories
                Subject: Case Report - Cyst and Tumors

                Keywords: epidermoid cyst,malignant transformation,squamous cell carcinoma,submandibular gland
                Data availability:
                Keywords: epidermoid cyst, malignant transformation, squamous cell carcinoma, submandibular gland

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