International Journal of Pediatric Otorhinolaryngology Extra
Volume 2, Issue 3 , Pages 176-179, September 2007

Infected dermoid cyst of the tongue with a sinus tract to the submental skin

  • Melanie Duval

      Affiliations

    • Department of Otolaryngology, McGill University, Montreal, Que., Canada
    • ,
  • Khalil Al-Macki

      Affiliations

    • Department of Otolaryngology, McGill University, Montreal, Que., Canada
    • ,
  • Moy-Fong Chen

      Affiliations

    • Department of Pathology, McGill University, Montreal, Que., Canada
    • ,
  • Peter J. Chauvin

      Affiliations

    • Department of Pathology, McGill University, Montreal, Que., Canada
    • ,
  • Ricardo Faingold

      Affiliations

    • Department of Radiology, McGill University, Montreal, Que., Canada
    • ,
  • John J. Manoukian

      Affiliations

    • Department of Otolaryngology, McGill University, Montreal, Que., Canada
    • Corresponding Author InformationCorresponding author at: 2300 Tupper Street, Montreal, Que. H3H 1P3, Canada. Tel.: +1 514 412 4304; fax: +1 514 412 4342.

Received 18 April 2007; received in revised form 13 May 2007; accepted 14 May 2007.

Article Outline

Summary 

Objectives: We report a case of a teratoid type dermoid cyst of the tongue with a sinus tract to the submental skin. Study design: Case report. Methods: We present a 3-month-old infant with a dermoid cyst of the tongue with a sinus tract to the submental skin and review of the literature on dermoid cysts of the tongue. Results: This rare case presented as an enlarging tongue mass and difficulty feeding. A midline submental skin pinpoint discharge revealed to be a sinus tract connecting the cyst to the skin. Complete excision of the cyst and sinus tract through midline ventral tongue and submental incisions was performed. Conclusion: Although dermoid cysts of the tongue are rare they should be part of the differential diagnosis of tongue masses and presence of a sinus tract should be considered in such cases.

Keywords: Dermoid cyst, Teratoid cyst, Tongue lesion, Tongue mass, Sinus tract, Infected dermoid cyst

 

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1. Introduction 

Dermoid cysts are uncommon lesions in the head and neck, with only 6.9% of dermoid cysts presenting in this region [1] and only 21 dermoid cysts of the tongue reported in the English literature. Of these 21 reported cases, 5 cases were of the teratoid subtype [2]. We report a case of intra-glossal teratoid type dermoid cyst with a cutaneous sinus tract to the submental skin.

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2. Case report 

A previously healthy 3-month-old infant presented to the emergency department with a 3-day history of intermittent high grade fever and poor feeding secondary to a swollen tongue. The parents had noticed the swelling 3 days ago and the tongue had been increasing in size since then, but was not causing any breathing difficulties. Physical examination revealed a healthy looking infant who was not in distress and had no dysmorphic features. The vital signs were all within normal limits with a temperature of 37°C, a blood pressure of 90/45mmHg and a heart rate of 145beats/min. The systemic examination was normal. Local examination revealed a tongue massively enlarged anteriorly and a midline submental pinpoint skin discoloration and discharge. Pertinent laboratory results included a white blood count of 17.6×109L−1. Computed tomography (CT) with contrast demonstrated a rim enhancing cystic lesion within the anterior two third of the tongue with some lobulation caudally, extending into the floor of the mouth and with a submental sinus tract. The differential diagnosis included an abscess or infected cyst with a sinus tract.

The patient was admitted to an observation unit with monitor and given IV 3rd generation cephalosporins (Ceftriaxone) and Decadron to reduce the swelling. Since the patient did not show any improvement and the swelling had increased after a 24h period, decision was made to proceed with surgery.

Surgery was performed to drain or remove the mass through a ventral midline tongue incision. Initially a midline intra-oral sublingual and ventral lingual approach was used to access the mass. Upon piercing of the capsule whitish purulence material was drained. The pus was sent for culture and sensitivity. An encapsulated mass with a 4cm sinus tract connecting the lingual mass to the submental skin was recovered A submental incision was also performed to free the sinus tract associated with the lesion. The mass and the sinus tract were excised completely and sent for pathological evaluation. Examination of the specimen revealed an infected intra-glossal dermoid cyst of the teratoma type with sinus tract lined by squamous and respiratory epithelium and containing adnexal structures. The infant was kept intubated for 24h post-operatively to let the inflammation settle and was continued on the same antibiotics. On follow-up visits 2 weeks and 2 months after the excision of the dermoid cyst the patient looked well and had no recurrence.

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3. Discussion 

Dermoid cysts are keratin-filled developmental cysts lined by stratified squamous epithelium and containing dermal appendages such as hair follicles, sebaceous and sweat glands. The term dermoid cyst is also used in a broader fashion to refer to epidermoid cysts which lack the adnexal structures and teratoid cysts which also contain mesodermal and endodermal elements.

The tongue is formed during the 4th week of gestation of two separate embryological entities, the anterior two third arising from the ectoderm and the posterior third from the endoderm [3]. The mucosal membrane of the anterior portion of the tongue is formed from the first branchial arch. Three swellings of primitive mesenchyme, the midline tuberculum impar and the bilateral lingual swellings, merge to form the anterior two third of the tongue. The mucosal membrane of the posterior third of the tongue is formed from a large midline swelling of mesoderm of the second, third and fourth branchial arches. The sulcus terminalis extending along the line of circumvalate papillae represents the junction of the anterior and posterior portions. The musculature of the tongue is formed 3 weeks later and originates from the migration of occipital somites.

Different hypothesis have been formulated on the origin of dermoid cysts. The most accepted one states that congenital lingual dermoid cysts arise from sequestration of dermal and epidermal cells during the midline fusion of the branchial arches [4]. A second hypothesis states that acquired dermoid cysts are formed by trapping of epidermal and dermal cells following trauma [5].

Sinus tract associated with dermoid cysts are not rare, at least six cases of dermoid cysts of the tongue associated with a drainage site or a sinus connecting to the cyst have been reported [6], [7], [8]. However, all reported sinus tracts were associated with true dermoid cysts [2] and the tracts were generally found on the dorsum of the tongue as opposed to a cutaneous fistula to the submental skin in our case. Dermoid cysts with a sinus tract presumably develop similarly to simple dermoid cysts, but connection to the surface is retained. A sinus tract could also develop following infection of the dermoid cyst [9]. In such a case, the sinus tract develops in the path of least resistance. The sinus tract allows drainage of the keratin and glandular products, but also makes the dermoid cyst prone to infections by entry of organisms from the skin or oral flora.

The presentation of most lingual dermoid cysts follows a bimodal distribution with the greatest peak during the teenage years and a smaller peak during the first year of life [10]. The cysts are generally quiescent until they become infected or so large that removal is required. Signs and symptoms include asymptomatic tongue enlargement, feeding difficulties and recurrent infections. The cyst can enlarge to a point that the patient is unable to close the mouth, chew or speak normally. Although this has never been reported for lingual dermoid cysts, there is a slight possibility of malignant transformation to squamous cell carcinoma [11]. The differential diagnosis of lesions of the tongue in a child should include enteric duplication cysts, hemangioma, lymphangioma, neurofibroma, salivary ranulas and ectopic thyroid [12] (Fig. 1, Fig. 2, Fig. 3, Fig. 4, Fig. 5, Fig. 6).

  • View full-size image.
  • Fig. 3. 

    Axial cut of the CT scan with contrast demonstrating a rim enhancing cystic lesion within the anterior two third of the tongue with lobulation caudally, extension into the floor of the mouth and a submental sinus tract.

  • View full-size image.
  • Fig. 4. 

    Saggital cut of the CT scan with contrast demonstrating a rim enhancing cystic lesion within the anterior two third of the tongue with lobulation caudally, extension into the floor of the mouth and a submental sinus tract.

Visualization of the dermoid cyst by CT or MRI helps to delineate the internal architecture and to reveal the position of the cyst in relation to the tongue musculature [7]. The CT with contrast material will typically show a unilocular mass with a well-defined enhanced capsule. The MRI characteristics are variable, the dermoid cyst being iso- or hypointense on T1-weighted images and hyperintense or heterogenous on T2-weighted images.

Definitive treatment consists of complete surgical removal, usually through an incision on the ventral surface of the tongue. Surgical excision entails the removal of the entire epithelial lining to avoid recurrence.

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4. Conclusion 

Although dermoid cysts are rarely found in the tongue, it should be included when formulating the differential diagnosis of a tongue mass, especially in infants and teenagers and in cases where a sinus tract is present. Complete excision of the cyst is necessary to avoid recurrence. This case illustrates the importance of considering the presence of a sinus tract which could either link the cyst to the oral cavity or the skin, as in our case.

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References 

  1. New GB, Erich JB. Dermoid cysts of the head and neck. Surg. Gynecol. Obstet. 1937;65:48–55
  2. Gleizal A, Abouchebel N, Lebreton F, Beiziat J-L. Dermoid cyst of the tongue: an association of dermoid cyst with bronchogenic epithelium. J. Craniomaxillofac. Surg. 2006;34:113–116
  3. Tomaski SM. Embryology and anatomy of the mouth, pharynx and esophagus. In: 3rd ed..  Bluestone CD,  Stool SE,  Kenna MA editor. Pediatric Otolaryngology. vol. 2:Philadelphia: W.B. Saunders; 1996;p. 918
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  5. Masliah MK, Blain S, Sanders B. Dermoid cysts of the oral regions in children. J. Pedod. 1979;3:221–234
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  8. Milam M, Hill SA, Manaligod JM. Lingual dermoid cysts. Otolaryngol. Head Neck Surg. 2003;128(3):428–429
  9. Shafer WG, Hine MK, Levy BM. A Textbook of Oral Pathology. 3rd ed.. Philadelphia: W.B. Saunders; 1974;p. 75
  10. Edwards CP, Lustrin L, Valderrama E. Dermoid cysts of the tongue: a review of five cases and review of the literature. Pediatr. Dev. Pathol. 2003;6:531–535
  11. Devine JC, Jones DC. Carcinomatous transformation of a sublingual dermoid cyst: a case report. Int. J. Oral Maxillofac. Surg. 2000;29:126–127
  12. Velcek FT, Klotz DH, Hill CH, Ladogana LE, Kottmeier PK. Tongue lesions in children. J. Pediatr. Surg. 1979;14:238–246

PII: S1871-4048(07)00042-1

doi:10.1016/j.pedex.2007.05.004

International Journal of Pediatric Otorhinolaryngology Extra
Volume 2, Issue 3 , Pages 176-179, September 2007

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