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DERMOID CYST OF THE ORAL CAVITY--A CASE REPORT/DERMOIDNA CISTA USNE DUPLJE--PRIKAZ SLUCAJA.

Introduction

Dermoid cysts are benign developmental growths that can be found in any part of the body. Cysts of the head and neck account for 7% of all cysts, and are most commonly located near the lateral aspect of the eyebrow. The second most common localization in the head and neck region is the floor of the mouth. Of all dermoid cysts, only 1.6% are located in the mouth, i. e. 23% of all cysts in the head and neck region. The floor of the mouth is the most common localization in the mouth, but rarely dermoid cysts are found in the tongue, lips, buccal mucosa, maxilla and mandible [1-3]. The origin of dermoid cysts still remains incompletely understood; they are considered to be congenital developmental abnormalities, with entrapment of pluripotent stem cells, implantation of the epithelial tissue, anomaly of thyroglossal duct or canal, or a consequence of a trauma. The most widely accepted theory is that cysts occur due to entrapment of ectodermal tissue in midline in third or fourth week of embryonic development, from the first or second branchial arch, therefore the term congenital cyst was accepted. They are commonly located in the midline. In 1955, Meyer classified the cysts of the oral cavity by histological features into three groups: epidermoid, dermoid and teratoid [1]. Dermoid cysts are layered by stratified squamous epithelium and contain skin and skin adnexa (glands and hair follicles). Epidermoid cysts are lined with stratified squamous epithelium and teratoid also contain skin adnexa, along with mesodermal and endodermal elements (muscles, bones, respiratory and gastrointestinal tissues) [1, 2, 4]. Dermoid and teratoid cysts may have a malignant potential [5].

These cysts are characterized by slow growth. They are most frequently found in children, in the second or third decade of life, equally in both genders, and are located in the midline [2]. These cysts may displace the tongue upwards and cause disturbances in speech, swallowing, and big cysts may cause airway obstruction. Sublingual localization is considered for cysts between the oral mucosa and geniohyoid muscles, submental most frequently between geniohyoid and mylohyoid muscles and outer between mylohyoid muscle and skin [4, 5]. The symptoms depend on the size and localization, but they frequently cause disturbances in speech and swallowing, or airway obstruction [1, 2, 5].

To confirm the clinical diagnosis and select a surgical strategy, it is necessary to perform imaging studies, ultrasound, computerized tomography or magnetic resonance imaging (MRI) [3, 6, 7].

The main therapeutical procedure is surgery, with intraoral approach for sublingual and submental cysts and external approach for cysts localized between mylohyoid muscles and skin [1, 2, 8].

Case report

A 15-year-old girl was referred to our Clinic due to a growth in the mouth, which was observed one year before. She complained of foreign body sensation and minimal disturbances in speech and swallowing. Her personal history showed a mitral valve prolapse.

Clinical examination: external inspection and palpation showed no pathological findings. During intraoral examination, intact mucosa was observed, with sublingual and submucosal growth in the midline. The body of the tongue was in the midline, motile, moderately displaced towards the palate. The mouth opening was within physiological limits. On palpation, a tumor in the body of the tongue was observed, with a diameter of 30 mm, with a soft, painless, elastic consistency. Laboratory findings were within the normal range.

The MRI of the floor of the mouth: in the midline, a clearly demarcated, oval, cystic lesion was observed, with dimensions of 31 x 29,5 x 19 mm, divided with a capsule from the surrounding muscles of the tongue, beyond the mylohyoid muscle, filled with liquid content, with homogenous hyperintense signal in T1W and T2W images. The airway was without pathological findings. On both sides, the jugular lymph node chains showed normal findings (Figure 1). After preoperative preparation, in general endotracheal anesthesia, using intraoral approach, typical for frenectomy, tumor excision was performed, with careful preparation of the lingual nerve and Wharton's duct, saving all blood vessels of the tongue, with blunt and sharp dissection. The tumor, adherent to the surrounding muscle tissues, was entirely dissected and extirpated. Macroscopically, it was a clearly demarcated tumor, with dimensions of 30 x 30 mm, of soft, elastic consistency. Preoperatively, the patient received antibiotic therapy (Ampicillin 2,0 i.v.). The postoperative course was uneventful, and on the fourth postoperative day the patient was discharged from hospital.

The cyst wall was lined with a stratified squamous epithelium and contained mature sebaceous glands. Keratin was also seen within the cyst cavity (HE, x 10). The histopathological findings suggested a dermoid cyst (Figure 2).

The patient was seen on regular follow-up visits, two years after surgery, having no subjective complaints and no clinical signs of recurrence.

Discussion

Dermoid cysts of the head and neck region account for 7% of all dermoid cysts. They are most frequently localized in the periorbital region [1, 3]. Other localizations are the floor of the mouth, submental or lateral submandibular region, forehead, neck and nose. The floor of the mouth is the most common localization for dermoid cysts of the mouth [1-3, 6]. Dermoid and epidermoid cysts of the mouth are rare lesions, i. e. from all dermoid cysts of the head and neck they account for 1-2%. They account for less than 0.01% of all cysts of the mouth. Dermoid cysts of the body of the tongue are very rare and in 90% of cases are found in childhood [3, 6]. Epidermoid cysts are layered with epithelium, while dermoid cysts contain skin adnexa and hair [2, 4, 5]. The origin of dermoid cysts of the mouth is dual. They are the consequence of the embryonic developmental disorder and entrapment of epithelial cells in the time of fusion of the first and second brachial arches, in the midline in the third and fourth embryonic weeks. Furthermore, dermoid cysts may occur due to iatrogenic damage or due to trauma, with epithelial and skin adnexa inclusion [4]. Dermoid cysts are most frequently diagnosed in the second and third decades of life, with no gender predominance, in midline, encapsulated [2]. Symptoms associated with dermoid cysts are dysphagia, dysphonia and speech problems, depending on their location and size. In our case, symptoms were moderate. A broad spectrum of lesions can be considered in differential diagnosis: infections, ranula, obstruction of the submandibular duct, tumors of the sublingual and other minor salivary glands, lymphadenopathy, thyroglossal duct cysts, lymphatic malformations, epidermoid cysts, neurofibromas, hemangiomas, heterotypic gastrointestinal cysts, foregut duplication cysts [1, 4, 5, 9]. The clinical findings are not sufficient. Valuable information can be gathered by ultrasound, but MRI is most important in the identification of the cysts, establishing the relationship with muscles of the floor of the mouth, and determining the surgical approach. The MRI findings of dermoid cysts have signals of variable intensity. Cysts can be hyperintense or isointense on T1-weighted images and usually the signal is of high intensity - hyperintense on T2-weighted images [3, 5, 6]. In our case report, like in other studies, dermoid cyst had shown a high intensity signal. Fine-needle aspiration biopsy is useful in making the diagnosis, but is not necessary [5]. The therapeutic surgical approach depends on the location of the cyst, anatomic localization and relationship with muscles of the floor of the mouth. In 1925, Colp classified dermoid cysts according to their relationship with the surrounding muscles as sublingual beyond the geniohyoid muscle, geniohyoid between geniohyoid and mylohyoid muscles and lateral beneath the mylohyoid muscle [8, 9]. Surgical excision is the only effective therapy. Most of the cysts beyond the mylohyoid muscle are appropriate for intraoral surgical approach, but with big cysts and those that are beneath the mylohyoid muscle--submandibular cysts, external approach is more appropriate [1]. In our case, the cyst was removed through intraoral incision approach, typical for frenectomy with precise dissection, which enabled thorough removal of the cyst. This approach allowed successful excision without or with minimal postoperative morbidity. A combined intra--and extraoral approach is reserved for cysts large in size [5]. Postoperatively, the attention should be paid to pain, edema, bleeding, troubles with breathing and swallowing, sensibility and function of the tongue, and on the course of healing [4]. It can be found in recent literature that endoscopic approach is suitable for growths in the medial upper region of the neck [8]. Provided that meticulous, thorough extirpation of the cyst is performed, no recurrences are found [3]. Most cysts already described in literature are of the same dimensions as in our case report [2, 5, 6, 8]. Rarely, they are larger [1, 3]. Definitive diagnosis is made by histological examination of the specimen. Dermoid cysts contain keratin, caseous contents, sweat and sebaceous glands, and hair follicles [4]. Malignant transformation of these cysts in squamocellular carcinoma is extremely rare, up to 5%, and was reported in case of sublingual dermoid cyst and in case of teratoid cyst of the floor of the mouth, after a long evolution process [3, 4, 10]. Radiation therapy is advised postoperatively.

Conclusion

Dermoid cysts in the region of the oral cavity are very rare; they are painless and grow slowly, but when they reach certain dimensions, they may disturb chewing, swallowing, and cause troubles with breathing. These cysts are benign lesions layered with epithelium which contains skin adnexa in the cyst wall. Dermoid cysts have a malignant potential. Magnetic resonance imaging provides complete information about the location, size, and content of the growth and contributes significantly to the decision about the surgical approach. Surgery is the only effective mode of therapy.

References

[1.] Aydin S, Demir MG, Demir N, Sahin S, Kayipmaz SS. A giant plunging sublingual dermoid cyst excised by intraoral approach. J Maxillofac Oral Surg. 2016;15(2):277-80.

[2.] Vieira EM, Borges AH, Volpato LE, Porto AN, Carvalhosa AA, Botelho Gde A, et al. Unusual dermoid cyst in oral cavity. Case Rep Pathol. 2014;2014:389752.

[3.] Shah SP, Quiroz F, Shah CJ. Dermoid cyst. Ultrasound Q. 2012;28(3):215-7.

[4.] Pirgousis P, Fernandes R. Giant submental dermoid cysts with near total obstruction of the oral cavity: report of 2 cases. J Oral Maxillofac Surg. 2011;69(2):532-5.

[5.] Papadogeorgakis N, Kalfarentzos EF, Vourlakou C, Alexandridis C. Surgical management of a large median dermoid cyst of the neck causing airway obstruction. A case report. Oral Maxillofac Surg. 2009;13(3):181-4.

[6.] Edwards PC, Lustrin L, Valderrama E. Dermoid cysts of the tongue: report of five cases and review of the literature. Pediatr Dev Pathol. 2003;6(6):531-5.

[7.] Kolomvos N, Theologie-Lygidakis N, Tzerbos F, Pavli M, Leventis M, Iatrou I. Surgical treatment of oral and facial soft tissue cystic lesions in children. A retrospective analysis of 60 consecutive cases with literature review. J Craniomaxillofac Surg. 2014;42(5):392-6.

[8.] Woo SH, Jeong HS, Kim JP, Park JJ, Baek CH. Endoscope-assisted frenotomy approach to median upper neck masses: clinical outcomes and safety (from a phase II clinical trial). Head Neck. 2014;36(7):985-91.

[9.] King RC, Smith BR, Burk JL. Dermoid cyst in the floor of the mouth. Review of the literature and case reports. Oral Surg Oral Med Oral Pathol. 1994;78(5):567-76.

[10.] Devine JC, Jones DC. Carcinomatous transformation of a sublingual dermoid cyst. A case report. Int J Oral Maxillofac Surg. 2000;29(2):126-7.

Rad je primljen 13. XI 2018.

Recenziran 17. XII 2018.

Prihvacen za stampu 10. I 2019.

BIBLID.0025-8105:(2018):LXXI:11-12:409-412.

University of Belgrade, School of Medicine, Belgrade (1)

Clinical Center of Serbia, Belgrade

Clinic of Otorhinolaryngology and Maxillofacial Surgery (2)

Milovan V. DIMITRIJEVIC (1,2), Ana M. DIMITRIJEVIC (1), Ivan V. BORICIC (1) and Petar M. DURKOVIC (1)

Corresponding Author: Prof. dr Milovan V. Dimitrijevic, Univerzitet u Beogradu, Medicinski fakultet, 11000 Beograd, Pasterova 2, Email: milovan.v.dimitrijevic@gmail.com

https://doi.org/10.2298/MPNS1812409D
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Title Annotation:CASE REPORT/PRIKAZ SLUCAJA
Author:Dimitrijevic, Milovan V.; Dimitrijevic, Ana M.; Boricic, Ivan V.; Durkovic, Petar M.
Publication:Medicinski Pregled
Date:Nov 1, 2018
Words:1975
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