Atormac
Home  -  About us  -  Editorial board  -  Search  -  Ahead of print  -  Current issue  -  Archives  -  Instructions  -  Subscribe  -  Contacts  -  Advertise - Login 
 
 
     

 Table of Contents  
CASE REPORT CYST & TUMORS
Year : 2020  |  Volume : 10  |  Issue : 1  |  Page : 258-261

Swelling in the floor of the mouth: A diagnostic dilemma


1 Department of ENT, MMDU, Ambala, Haryana, India
2 Department of Head and Neck Surgery, MMDU, Ambala, Haryana, India

Date of Submission03-Dec-2019
Date of Acceptance07-Feb-2020
Date of Web Publication8-Jun-2020

Correspondence Address:
Dr. Ankita Yadav
D-102 The Villas, DLF Phase 2, Gurugram - 122 002, Haryana
India
Login to access the Email id


DOI: 10.4103/ams.ams_263_19

Rights and Permissions
  Abstract 


Epidermoid cysts are abnormal structures originating from abnormal ectoderm and are benign and slow growing. These are found anywhere in the body. They arise during embryonic fusion due to entrapment of epithelial remnants and sometimes due to faulty implantation of the epithelium. The cysts can be epidermoid (squamous epithelium), true dermoid (skin apppendages), and teratoid (all germ layers). They are usually diagnosed by fine-needle cytological aspiration and require imaging to plan line of treatment. Diagnostic dilemma is always an issue as cystic lesions are commonly found in the head-and-neck region. Here, we present a case of large epidermoid cyst involving sublingual and submandibular region.

Keywords: Epidermoid cyst, fine needle aspiration cytology, plunging ranula


How to cite this article:
Datta G, Yadav A. Swelling in the floor of the mouth: A diagnostic dilemma. Ann Maxillofac Surg 2020;10:258-61

How to cite this URL:
Datta G, Yadav A. Swelling in the floor of the mouth: A diagnostic dilemma. Ann Maxillofac Surg [serial online] 2020 [cited 2020 Sep 25];10:258-61. Available from: http://www.amsjournal.com/text.asp?2020/10/1/258/286168




  Introduction Top


Epidermoid, dermoid and teratoid cysts are cystic malformations with squamous epithelium lining. They are classified as epidermoid (squamous epithelium), dermoid (skin adnexa), and teratoid (tissue as muscle, cartilage, and bone).[1],[2] They are gradually progressing, benign lesions, which arise from abnormal ectoderm. A simple cyst is lined with squamous epithelium and has no other adnexal structures with or without keratinous material. Epidermal cysts could be found anywhere in the body, head and neck (7%) and oral cavity (1.6%).[3],[4],[5] The most common site is the floor of the mouth, followed by the lips, tongue, and buccal mucosa.[6],[7] Diagnosis is made by imaging assisted by fine-needle aspiration and excisional biopsy. The head-and-neck region is a common site for different types of clinically similar and comparable lesions, hence making diagnosis difficult, thus complicating decision making regarding the line of treatment.

Epidermoid cysts present with male predilection, most commonly seen in the age group of 15 and 35 years but can present in any age group. They are gradually progressive benign masses, which can cause pressure symptoms such as dysphagia and dyspnea when large in size.[3] The patient can present with unusual symptoms such as pain, respiratory distress and speech disorder when involving the lower lip, upper lip, or oral cavity.[6] Head-and-neck is an unusual site for epidermoid cysts thus having a high chance for misdiagnosis. Here, we discuss managing strategies and problems faced while treating a case of an epidermoid cyst which was misleading in its clinical and radiographical profile.


  Case Report Top


A 24-year-old male patient presented to the outpatient department with a right-sided neck swelling for 8–9 months. The swelling was insidious in onset and gradually progressive in nature, started as the size of a pea and progressed to present size. He noticed a swelling on the right side floor of the mouth after a period of 2 months from the initial swelling in the neck. He gave no history of pain, discharge from swelling, difficulty swallowing, difficulty breathing, and no association with fever. Medical and dental history was not significant.

On examination

Extraorally swelling was found in the right submandibular region, round in shape measuring approximately 5 cm × 3 cm in dimension [Figure 1]. On palpation, it was nontender without local rise in temperature, freely mobile, and no fixity were observed to the underlying structure. On inspection, the skin was normal in color and texture and was pinchable.
Figure 1: Extraoral swelling

Click here to view


Intraoral examination revealed a swelling on the right side floor of the mouth not crossing midline measuring approximately 3 cm × 1 cm. The swelling was cystic in consistency, fluctuant, compressible, and nontender; tongue mobility was normal.

Fine-needle aspiration cytology (FNAC) of the submandibular swelling was done; findings were suggestive of a sebaceous cyst. Ultrasonography (USG) feature of the neck was suggestive of a dermoid cyst.

Contrast-enhanced computed tomography (CECT) of the neck [Figure 2]a,[Figure 2]b, [Figure 2]c showed a large nonenhancing well-circumscribed cystic lesion in the floor of the mouth involving right sublingual space and extending into the right submandibular space with the presentation of a plunging ranula.
Figure 2: (a) Intraoral swelling, (b) Axial view showing nonenhancing mass, (c) Sagittal view nonenhancing right submandibular, sublingual region

Click here to view


Enucleation of the cyst was done by a submandibular approach with the removal of the involved submandibular gland under general anesthesia, along with an intraoral approach removing all adhesions along the lesion and removing sublingual gland delivered through a submandibular approach [Figure 3].
Figure 3: Intraoperatively showing two cystic swellings delivered via the submandibular approach

Click here to view


The specimen received measured 3 cm × 2.5 cm in dimension [Figure 4], yellow in color, and doughy consistency on palpation. Gross examination showed cheesy material inside with a thin external capsule [Figure 5].
Figure 4: Specimen measuring 3 cm × 2 cm

Click here to view
Figure 5: Cheesy material inside the swelling

Click here to view


On histopathological examination

Chronic inflammatory cells were seen in the cystic lining and connective tissue capsule under high-power microscope (×40), with staining hematoxylin and eosin (H and E) [Figure 6].
Figure 6: Histopathological slides

Click here to view


A hematoxylin and Eosin stained section of the cyst under 4x magnification was seen to be lined by keratinized stratified squamous epithelium, with an excess amount of keratin in the lumen. Chronic inflammatory cells were found in connective-tissue capsule (no skin appendages visualized) [Figure 7]. Histopathologcal examination was suggestive of the dermoid cyst.
Figure 7: Histopathological slides

Click here to view
Figure 8: Post operative picture showing the incision with drain in situ

Click here to view



  Discussion Top


Neck masses are a common presentation to an otorhinolaryngologist. In the adult population, 80% of nonthyroid masses are neoplastic. Of these, 75% account for metastatic masses. In children below 15 years, 90% of masses are benign out of which 55% are congenital. Epidermoid cysts in the form of cutaneous lesions most commonly occur in the scalp, face, and trunk. As seen in older and in recent literature, epidermoid cysts and dermoid cysts in the deeper planes are less common and account for only 1.6-6.9%.[3] In a retrospective study of 89 children, a majority of 58.88% cases were dermoid cyst and only 13.33% were epidermoid cyst.[7] In a study conducted by New and Erich at the Mayo clinic on 1495 cases of dermoid cysts, only 24 were intraoral, in the floor of the mouth and most commonly reported in ovaries and testicles (80%) and about 7% seen in head and neck.[8],[9] They are quite uncommon in the oral cavity, comprising <0.01% of all oral cysts.[9],[10],[11] Epidermoid cysts are benign cysts seen anywhere in the body, most commonly seen at embryonic fusion sites.

Epidermoid cysts in the floor of the mouth occur due to epithelial remnants getting entrapped in the course of midline closure of the second and third branchial arches during 3rd–5th week of gestation. It can also be due to ectodermal differentiation of multipotent cells which have been pinched off during anterior neuropore closure or can be a thyroglossal duct cyst variant. Irrespective of the etiology, such cystic swellings remain unnoticed until it produces a mechanical obstruction, as seen in our patient. Epidermoid cysts can be of two types based on their origin, i.e., congenital or acquired, but both are the same clinically as well as histopathologically. Age group is no bar but mainly seen between 15 and 35 years of age with male predominance. Although most common site here is the midline of the floor of cavity, occasionally involving the buccal mucosa, lips, tongue, uvula, temporomandibular joint dermal graft, etc., because of the above mentioned, mylohyoid muscles pushes the tongue which causes symptoms such as dysphagia, dyspnea, and dysphonia, while below it causes swelling below the chin.

Epidermoid cysts on palpation have dough-like consistency or fluctuant and cyst-like consistency, with contents being cheesy to the liquefied substance with cyst wall of 2–6 mm thickness,[5] as clearly seen in our patient. In confirming the diagnosis and classification of cyst imaging modalities play a key role. The initial line of investigation is USG. In our patient, USG was suggestive of dermoid cyst, whereas fine-needle aspiration was suggestive of sebaceous cyst. The head-and-neck region swelling presents with wide range of differential diagnosis. To confirm the diagnosis, we planned a CECT scan for the patient. Epidermoid cysts are well-defined cyst with multiple echogenic nodules in the cyst. CECT scan gives an appearance of the unilocular cyst with hypoattenuating homogeneous cyst with multiple hypoattenuating fat densities giving a sack of marble appearance. The CECT scan in our patient was suggesting of plunging ranula which added to the confusion in confirming the diagnosis. CECT helped in differentiating vascular lesions such as capillary hemangioma, arterial malformation, and venous malformation. Treatment comprises total surgical excision, the approach is decided based on the site of the cyst with respect to the muscle plane in the floor of the mouth either intraorally (small sublingual lesion above the mylohyoid muscle) or externally (cervical approach for large submandibular or submental lesions below the geniohyoid).[3] A combined approach (intraoral & cervical) may be considered in large lesions, typically presenting as dumbbell. The computed tomography (CT) imaging of the patient did not clearly guided us for the approach to take as it was a dumbbell-shaped cyst occupying the floor of the mouth and submandibular gland. Clinically and radiologically, we were in favor of plunging ranula, as it is more commonly encountered entity than an epidermoid cyst.


  Conclusion Top


Epidermoid cysts of the head-and-neck origin are rare findings and hence likely to be misdiagnosed. The chances of misdiagnosis are manifold due to unusual and misleading clinical presentation. We have found preoperative FNAC more informative and guiding. CT scan is a necessity to guide the anatomical plane, nature of the surgery, and plan surgery. Here, we present a simple case of a epidermoid cyst which was initially diagnosed as lipoma, FNAC was suggestive of a dermoid cyst, and CT suggestive of plunging ranula later turned out to be an epidermoid cyst, so the varied and extraordinary presentation of epidermoid cysts is challenging and entertaining for the otorhinolaryngology clinicians.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
De Ponte FS, Brunelli A, Marchetti E, Bottini DJ. Sublingual epidermoid cyst. J Craniofac Surg 2002;13:308-10.  Back to cited text no. 1
    
2.
Meyer I. Dermoid cysts (dermoids) of the floor of the mouth. Oral Surg Oral Med Oral Pathol 1955;8:1149-64.  Back to cited text no. 2
    
3.
Jham BC, Duraes GV, Jham AC, Santos CR. Epidermoid cyst of the floor of the mouth: A case report. J Can Dent Assoc 2007;73:525-8.  Back to cited text no. 3
    
4.
Pereira CM, Gasparetto PF, de Lucena Botelho T. Atypical appearance of epidermoid cyst in tongue's ventral surface. RSBO 2011;8:240-2.  Back to cited text no. 4
    
5.
Mirza S, Fadl S, Napaki S, Abualruz A. Case report of complicated epidermoid cyst of the floor of the mouth: Radiology-histopathology correlation. Qatar Med J 2014;2014:12-6.  Back to cited text no. 5
    
6.
Handa U, Kumar S, Mohan H. Aspiration cytology of epidermoid cyst of terminal phalanx. Diagn Cytopathol 2002;26:266-7.  Back to cited text no. 6
    
7.
Armon N, Shamay S, Maly A, Margulis A. Occurrence and characteristics of head cysts in children. Eplasty 2010;10:e37.  Back to cited text no. 7
    
8.
Koca H, Seckin T, Sipahi A, Kazanc A. Epidermoid cyst in the floor of the mouth: Report of a case. Quintessence Int 2007;38:473-7.  Back to cited text no. 8
    
9.
Ozan F, Polat HB, Ay S, Goze F. Epidermoid cyst of the buccal mucosa: A case report. J Contemp Dent Pract 2007;8:90-6.  Back to cited text no. 9
    
10.
Pancholi A, Raniga S, Vohra PA, Vaidya V. Midline submental epidermoid cyst: A rare case. Internet J Otorhinolaryngol 2006;4:1-5.  Back to cited text no. 10
    
11.
Kandogan T, Koç M, Vardar E, Selek E, Sezgin O. Sublingual epidermoid cyst: A case report. J Med Case Rep 2007;1:87.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]



 

Top
 
 
Search
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)  

 
  In this article
   Abstract
  Introduction
  Case Report
  Discussion
  Conclusion
   References
   Article Figures

 Article Access Statistics
    Viewed204    
    Printed14    
    Emailed0    
    PDF Downloaded43    
    Comments [Add]    

Recommend this journal