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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 14  |  Issue : 2  |  Page : 96-100

Sublingual dermoid cyst: Case series


Department of Oral and Maxillofacial Surgery, Army Dental Centre R and R, New Delhi, India

Date of Submission22-May-2020
Date of Acceptance23-May-2020
Date of Web Publication15-Jul-2020

Correspondence Address:
Sudarshan Bhat
Department of Oral and Maxillofacial Surgery, Army Dental Centre R and R, New Delhi - 110 010
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JODD.JODD_37_20

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  Abstract 


Dermoid cysts in the floor of the mouth may be congenital or acquired. The congenital form originates from embryonic cells of the 1st and 2nd branchial arch. The acquired form may be due to traumatic or iatrogenic causes. Its occurrence is estimated to be from 1.6% to 6.9% of the dermoid cysts of the body in adults. They may be classified anatomically and histologically. Anatomically, based on the location while histologically, they are divided into epidermoid, dermoid cysts, and teratomas. Enucleation through intraoral and/or extraoral approach is the method of treatment. A case series of dermoid cysts in the floor of the mouth are presented in this article; and an evaluation with regard to pathology, clinical findings, and treatment is discussed.

Keywords: Dermoid cyst, floor of the mouth, sublingual dermoid


How to cite this article:
Sahoo N K, Roy I D, Sharma R, Saxena V, N Babu B K, Bhat S. Sublingual dermoid cyst: Case series. J Dent Def Sect. 2020;14:96-100

How to cite this URL:
Sahoo N K, Roy I D, Sharma R, Saxena V, N Babu B K, Bhat S. Sublingual dermoid cyst: Case series. J Dent Def Sect. [serial online] 2020 [cited 2020 Aug 5];14:96-100. Available from: http://www.journaldds.org/text.asp?2020/14/2/96/289752




  Introduction Top


Dermoid cysts are considered to be developmental anomalies, and they are commonly found in the anal region, testis and ovaries.[1],[2] These cysts are rarely found in the maxillofacial region. They constitute 1.6%–6.9% of all cysts in the head and neck area and are most commonly found around the orbit and the nasal complex.[3] The other rarely occurring sites are occipital, frontal, and labial region. The etiology of these lesions is not well established, but several theories have been proposed, the most accepted theory suggests that the dermoid cyst of floor of the mouth may be congenital or acquired. The congenital form originates from embryonic cells of the mandibular and hyoid branchial arches. The acquired form may be due to traumatic or iatrogenic cause. Mostly, these lesions occur as slowly enlarging masses in young adults.[1] Surgical removal is the treatment of choice for dermoid cysts found in floor of the mouth. Here, we present a case series of dermoid cysts in the floor of the mouth which were treated by total surgical excision.


  Case Reports Top


Case-1

A 16-year-old male reported to our center with a chief complaint of a swelling on his chin and tongue. At 6 years of age, his mother had taken him to a physician for the same and no active was carried out. Due to the gradual increase in size of the swelling, double-chin appearance and some difficulty in pronouncing certain words he has reported for consultation. Extraoral examination revealed an obvious mid-line submental swelling of approximately 5 cm × 3 cm in size [Figure 1]. The margins were irregular, surface was smooth, and the overlying skin was normal. On palpation it was nontender, compressible, doughy in consistency, nonpulsatile and not fixed to the underlying structures. There was no lymphadenopathy. Temporomandibular joints were normal, and mouth opening was adequate. Floor of the mouth and tongue were slightly raised. On digital pressure over the submental area, the swelling protruded into the sublingual area. Tongue movements were mildly restricted. The oral mucosa and Wharton's ducts were healthy bilaterally. Some movement of the swelling was observed during swallowing. A clinical diagnosis of sublingual dermoid cyst was made, and plunging ranula was considered in the differential diagnosis.
Figure 1: Swelling below the chin

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He was investigated by magnetic resonance imaging (MRI) which showed a hypointense and semisolid mass with a well-defined capsule originating from the anterior two thirds of the digastric muscle groups on T1-weighted images. The sagittal and coronal sections [Figure 2] showed a multilocular lesion filling the entire floor of the mouth. The lesion was extending above and below the mylohyoid muscle and anteriorly to the lingual surface of the mandible. The posterior border of the lesion was defined by displaced intrinsic muscles of the tongue.
Figure 2: Magnetic resonance imaging sagittal section

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Aspiration was carried out with a wide bore needle inserted into the most prominent submental aspect of the swelling. About 1 cc of thick, granular, yellowish fluid was aspirated. Under the microscope, these fine granules were seen to consist of clumps of cholesterol crystals.

Surgical excision was carried out under general anesthesia (GA) through a combination of intraoral and extraoral approach. The intra oral incision was placed lateral and parallel to the Wharton's duct, and the lesion was exposed. The dissection was carried to the level of the genial tubercles and the mylohyoid ridge and decompressed by aspirating some of the cystic content to avoid rupture. Extraorally, the lesion was exposed though submental approach. The anterior bellies of the digastric muscles were retracted laterally to gain access to the superior component of the cyst. The mass was freed from surrounding tissue and enucleated [Figure 3]. The wound was irrigated; vacuum drain placed and closed in layers. Patency of the sumandibular ducts was verified. The postoperative healing was uneventful.
Figure 3: Enucleated specimen

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The specimen was sent for histopathological examination which showed stratified squamous epithelial lining with keratin filaments and sebaceous glands [Figure 4]. The diagnosis of dermoid cyst was confirmed.
Figure 4: Microscopic view showing stratified squamous epithelial lining with keratin filaments and sebaceous glands

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Case-2

A 21-year-old male reported to our center with a painless slow-growing swelling over the chin for last 10 years. Earlier, he has consulted a general practitioner for the same without much relief. Extra oral examination revealed a painless, nontender, soft, smooth mass having doughy consistency measuring around 5 cm × 4 cm that extended from the submental to the right submandibular region [Figure 5]. It was compressible, nonpulsatile and was not fixed to the underlying structures. There was no lymphadenopathy. The overlying skin was normal. Intraoral examination was unremarkable.
Figure 5: Swelling in the submandibular region

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MRI revealed a well-circumscribed unilocular mass with intermediate signal intensity and an homogeneous internal structure in the submental area extending to right submandibular region. The sagittal and coronal sections showed a unilocular lesion below the mylohyoid muscle. A yellowish-white paste-like material was aspirated from the lesion. Cytological examination was negative for atypia and confirmed the contents of a dermoid cyst.

Surgical excision of the lesion was carried out under GA through extraoral approach. The lesion was exposed completely by blunt dissection without rupturing the fibrous capsule. The cystic fluid was aspirated, and the lesion was completely enucleated in toto [Figure 6]. Postoperative healing was uneventful. Microscopic examination revealed a lumen lined with stratified squamous epithelium with a layer of orthokeratin and a connective tissue capsule with sebaceous glands. The diagnosis of a dermoid cyst was confirmed.
Figure 6: Enucleated specimen

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Case-3

A 24-year-old female patient reported to our department with complaint of slowly enlarging painless swelling below the chin for 3 years. The patient gave a history of surgical excision of ranula in the sublingual region when she was 1 month old.

On extraoral examination, a solitary, well demarcated, nontender, nonpulsatile, midline swelling of approximately 4 cm × 5 cm in size, having doughy consistency was noticed in the submental region [Figure 7]. Mouth opening was adequate, and there was no associated inflammatory sign or lymphadenopathy. Mild swelling was also observed intra orally in the sublingual region. Tongue movement was intact. Sagittal and coronal view of MRI showed a large, well circumscribed medially located cystic mass of size 35 mm × 40 mm in size. Aspiration was done, a thick yellowish cheesy material was aspirated and cytologic examination confirmed the contents of a dermoid cyst. After obtaining informed consent, individual was taken up for surgical excision of the lesion through intraoral approach under GA with nasotracheal intubation.
Figure 7: Swelling below the chin

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Intraoperatively, a midline incision from the base of tongue to the floor of mouth was done using electrocautery to access the lesion. Wharton's ducts were identified and protected. The cystic mass was dissected carefully from surrounding tissue using sharp and blunt dissection. The geniohyoid muscle was dissected, and enucleation was done [Figure 8]. The soft-tissue specimen was sent for histopathological examination which confirmed the diagnosis of a dermoid cyst. Postoperative recovery was uneventful. The patient is under follow-up for 1 year with no signs of recurrence.
Figure 8: Enucleated specimen

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  Discussion Top


Dermoid cysts of the oral floor are an uncommon entity, but when they occur, they usually manifest during the second or third decades of life with no gender predilection.[4] It may occur as cystic, slightly yellowish mass in the midline of floor of the mouth or as a swelling below the chin; their most frequent location is in or around the midline in the floor of the mouth. Occasional occurrence involving the buccal mucosa, tongue, lips, uvula, temporomandibular joint, intradiploic, intracranial, and intraosseous location within the mandible and maxilla also have been reported.[5] In our case, all the patients were aged below 25 years and had an extraoral swelling in the submandibular region. In addition to the extraoral swelling, case 1 and 3 reported to us with an extension of the swelling in the sublingual region also. Due to the upward displacement of the tongue, symptoms of dysphagia, dyspnea, and dysphonia may occur in severe cases. Our first case had mild restriction of tongue movements with difficulty in pronouncing certain words due to a raised tongue. These cystic swellings may grow inferiorly and may give rise to the characteristic “double-chin” appearance,[6] which was evident in all our cases. Dermoid cysts on palpation typically feel “dough like”, although they may be fluctuant depending upon the consistency of the luminal contents that may range from a cheesy, sebaceous to liquefied substance.

Ultrasonography (USG), computed tomography (CT), and MRI are some of the diagnostic tools which help in better preoperative planning. USG reveals either a solid or cystic structure within a heterogeneous mass. CT scans reveal the pathognomonic “sack-of marbles” appearance due to hypoattenuating fat nodules.[4] MRI of dermoid cysts give variable signal intensity on T1-weighted images and are usually hyperintense on T2-weighted images these findings are of considerable importance in depicting the relationship of cystic mass and muscles of floor of the mouth. Based on the anatomical location of the lesion, Teszler et al. have classified the dermoid cysts as supramylohyoid (intraoral or sublingual), inframylohyoid (Cervical) and peri- and trans-mylohyoid (dual intraoral and cervical).[7] In all our cases, MRI was done. In case 1, the lesion was perimylohyoid and trans-mylohyoid; in case 2, it was inframylohyoid; and in case 3, it was supramylohyoid.

Aspiration with a wide bore needle was carried out in all the patients, which revealed a thick, granular, yellowish paste like fluid containing numerous fine yellow granules which are the characteristics of dermoid cyst. The differential diagnosis includes ranula, thyroglossal duct cyst, cystic hygroma, branchial cleft cysts, and benign and malignant tumors of the floor of the mouth and adjacent salivary glands.

The treatment advocated for dermoid cyst is total surgical excision. Surgical approach depends upon site and size of the lesion. Intaoral approach, for cystic mass situated above the mylohyoid muscle and submandibular approach, if the cystic mass is present below the mylohyoid muscle. Large peri- and trans-mylohyoid cysts may require both intraoral and extraoral incisions to provide the direct visualization of important adjacent structures which was done in our 1st case. In case 2, as it was arising in the submandibular region on the right side, it could be most conveniently excised through a submandibular approach, and in case 3, as the cystic mass was present above the mylohyoid muscle an intraoral approach was preferred. Care should be taken not to rupture the cyst, as cystic contents may act as irritants to fibrovascular tissues, causing postoperative inflammation. The surgical site should be irrigated thoroughly before the closure.

The generic term “dermoid cyst” or “dermoid” was used to describe three histologic varieties: epidermoid cyst, true dermoid cyst, and teratoid cyst. The distinction between these cysts also carries odontogenic and oncologic elements. All three types may contain a cheesy keratinous material.[8] In all of our cases, the histopathological examination of the specimen confirmed the diagnosis of true dermoid cyst. Histopathologically, an epidermoid cyst consists of simple squamous epithelium with a fibrous wall, whereas a dermoid cyst in addition has varying number of skin appendages such as hair follicles and sebaceous glands. This is also known as a compound cyst.[5],[9] Teratoid cyst is lined with a range of epithelia from simple squamous epithelium to ciliated respiratory type, containing derivatives of ectoderm, mesoderm, and endoderm. This is also known as a complex cyst.[5],[9] There are the reports of malignant transformation of sublingual dermoid and epidermoid to squamous carcinoma and basal cell carcinoma. A 5% rate of malignant transformation of the teratoid variety of oral dermoid cysts has been reported.[10] Recurrence is unusual after complete surgical excision, all our cases are being followed up every 6 months, and there has been no evidence of recurrence.


  Conclusion Top


Dermoid cysts of the floor of the oral cavity are rare. When they occur, they may interfere with deglutition, phonation, and may also lead to respiratory obstruction. Early diagnosis and treatment are essential for these cystic entities. Appropriate imaging techniques are necessary in the preoperative diagnosis of cysts of the floor of the mouth. Surgical excision is the ideal treatment for such lesions. In view of recurrence, long-term follow-up is recommended.

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.
Zeltser R, Milhem I, Azaz B, Hasson O. Dermoid cysts of floor of the mouth: Report of four cases. Am J Otolaryngol 2000;21:55-60.  Back to cited text no. 1
    
2.
Longo F, Maremonti P, Mangone GM, De Maria G, Califano L. Midline (dermoid) cysts of the floor of the mouth: Report of 16 cases and review of surgical techniques. Plast Reconstr Surg 2003;112:1560-5.  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.
Sahoo NK, Choudhary AK, Srinivas V, Kapil T. Dermoid cysts of maxillofacial region. Med J Armed Forces India 2015;71:389-94.  Back to cited text no. 4
    
5.
Shear M, Speight P. From developmental cysts of head and neck. In: Cysts of Oral and Maxillofacial Region. 4th ed. Singapore: Blackwell Munksgaard; 2007. p. 181-3.  Back to cited text no. 5
    
6.
Kandogan T, Koc 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. 6
    
7.
Teszler CB, El-Naaj IA, Emodi O, Luntz M, Peled M. Dermoid cysts of the lateral floor of the mouth: A comprehensive anatomo-surgical classification of cysts of the oral floor. J Oral Maxillofac Surg 2007;65:327-32.  Back to cited text no. 7
    
8.
De Ponte FS, Brunelli A, Marchetti E, Bottini DJ. Sublingual epidermoid cyst. J Craniofac Surg 2002;13:308-10.  Back to cited text no. 8
    
9.
Koca H, Seckin T, Sipahi A, Kaznac A. Epidermoid cyst in the floorof the mouth: Report of a case. Quintessence Int 2007;38:473-7.  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;4:74-7.  Back to cited text no. 10
    


    Figures

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



 

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