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Case ReportExtensive Epidermoid Cyst and Breathing Difficulty

Ciro Dantas Soares,1 Alberto Costa Gurgel,2

Francisco de Assis de Souza Jnior,2 Samila Neres de Oliveira,2

Maria Goretti Freire de Carvalho,2 and Hanieri Gustavo Oliveira2

Department of Oral Diagnosis, Piracicaba Dental School, Universidade Estadual de Campinas (UNICAMP),Avenida Limeira , P.O. Box , Piracicaba, SP, BrazilDepartment of Dentistry, Universidade Potiguar (UnP), Avenida Senador Salgado Filho , - Natal, RN, Brazil

Correspondence should be addressed to Ciro Dantas Soares; [email protected]

Received March ; Accepted May

Academic Editor: Yuk-Kwan Chen

Copyright Ciro Dantas Soares et al. Tis is an open access article distributed under the Creative Commons AttributionLicense, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properlycited.

Epidermoid cysts are common cystic lesions in the skin, ovaries, and testicles, but their occurrence in the oral cavity is uncommon.Tey consist o cysts delimitedby a brous capsule without cutaneous annexes andare lined by stratied squamous epithelium. Tedifferential diagnosis includes ranula, dermoid cysts, and lingual thyroid. Despite their benign presentation, these cysts can causeunctional limitations, requiring special clinical attention or extensive lesions located in regions that preserve vital structures. Tis

paper aims to report a case o epidermoid cyst in patient with swallowing and breathing difficulty, highlighting the clinical andsurgical planning.

1. Introduction

Epidermoid cysts (epidermic cysts, EC) are common cysticlesions in skin, testicles, and ovaries derivates o ectoderm-lined inclusion. Tey comprise less than .% o all oralcavity cysts and their occurrence in the oor o the mouthwith respiratory complications has not been reported [,].

Histologically, EC are bounded by brous capsule and arecomposed o an epithelium which is attened and contains

a granular layer o keratohyalin granules. Absence o hairollicles, sebaceous glands, and apocrine sweat glands (skinappendages) in capsule o these cysts helps differentiatebetween dermoid cysts []. Te dermoid and epidermoidcysts are indistinguishable in the clinical and radiographicexams and require microscopic analysis or differentiation[].

Etiology o EC remains unknown and the most acceptedtheory is the reactivation o the remaining ectoderm trappedin the st and nd pharyngeal arches. However, accidental ortraumatic inclusion o epithelial tissue in deep structures othe dermis or submucosa may be associated with pathogene-sis o epidermoid cysts [,].

Te epithelial cells malignant transormation o thesecysts has been reported but is rare [,]. Extensive lesionslocated at regions that preserve vital structures may causeunctional limitations, requiring special clinical attention.Early diagnosis o epidermoid cysts permits good unctionaland aesthetic results. Te need or interaction in a multidis-

ciplinary team must be assessed [,].

Tis paper aims to report a case o an extensive epider-

moid cyst on buccal oor, with emphasis on the importanceo image diagnosis (Cone-Beam Computed omography) ortreatment planning.

2. Case Report

A -year-old male patient presents with extensive massin the buccal oor, with limitation in mouth opening andspeech associated with dysphagia and dyspnea. Te period oevolution o lesion was unknown. Te clinical examinationrevealed an expansive mass, asymptomatic, exophytic, andno history o associated trauma, and uctuated upon pal-pation (Figure ). Te lesion surace had normal-appearing

Hindawi Publishing CorporationCase Reports in DentistryVolume 2015, Article ID 826389, 4 pageshttp://dx.doi.org/10.1155/2015/826389


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F : Clinical aspect o mass in oor o the mouth, asymp-tomatic. Te patient related difficulty breathing and swallowing.

overlying mucosa. Te clinical diagnosis was ranula, dermoidcyst, or epidermoid cyst.

As patient reported swallowing and breathing difficulty,additional hematological examinations were perormed,

which showed normal range values. For surgery planningpurposes and or observed relationship with sof tissuesand other anatomical structures, a C scan was perormedshowing the dimensions o the lesion, as well as conrmingthe hypothetical diagnosis o the internal liquid contents.Aspiration puncture demonstrated content material riableand white. Surgical planning included complete lesion exci-sion. Afer this the specimens were removed and were sentor anatomical pathologic evaluation.

Te ovoid cystic mass was macroscopically observed tobe opened and without any content. It was measured to be. . . cm and it had brown pigmentation with aew whitish areas. Microscopic examination revealed a cystic

cavity with a capsule composed o dense brous connectivetissue, lined by stratied squamous epithelium resemblingepidermis (Figure (a)). Tere were no skin appendages inthe capsule. Te lesion contents were represented by concen-tric blades o orthokeratin. A breach on the cyst wall withchronic granulomatous inammation and multinucleatedgiant cells was also observed (Figure (b)), including keratin,being the nal diagnosis o a ruptured epidermoid cyst, withgranuloma to the oreign body (keratin).

3. Discussion

Te epidermoid cysts (EC) have uncertain etiology and

may be ormed rom reactivation o epithelial remnantsentrapped during midline closure o the bilateral rst andsecond branchialarches. Another probablecause is accidentalintroduction o epithelium in the subcutaneous tissues or inthe submucosa afer extraction o a third molar, or example,[,,].

Tey affect mainly male patients and are uncommonduring childhood or puberty. Teir occurrence in the oralcavity is rare and represents less than .% o all oral cysts[,]. Despite the benign course o EC, these lesions mayreach masses o large volume due to production o keratinwithin the cyst, as an attempt to balance the osmotic pressure,and were related to its malignant transormation [,].

F : Cone-Beam Computed omography. Axial section show-ing extensive lesion on the buccal oor (in the sublingual space),demonstrating hypodense areas and almost airway obstruction (redarrows).

F : Aspect o contents o lesion during surgical excision:material white, riable, and compatible with orthokeratin.

EC have different diagnosis such as inectious lesionso the salivary glands, ranula, dermoid cyst, lipoma, lingualthyroid, and thyroglossal duct cyst [, , , ]. In thepresent case the hypothesis o diagnosis was ranula, dermoidcyst, and EC (Figure ). Te patient reported swallowingand breathing difficulty, probably occasioned or posteriorexpansion o the lesion in submandibular space.

Computed tomography (C) is a reliable assessment olesion extension to deeper structures when diagnosing orevaluating the submandibular space. C permits visualiza-tion o the differences in densities o hard and sof tissuesthus optimizing the diagnosis and it guides the surgeon ora more efficient treatment plan and enables visualizing therelationship o the lesion with muscles, salivary glands, andother tissues [, , ]. Te submandibular space containssubmandibular salivary gland; and the lingual nerve, artery,andvein andthese structures are necessary or the physiologyo oral cavity.

In this case or sae surgical approach,was observedthe depth o the lesion in the submandibular space. Alsoit became evident that the lumen o the lesion is o lowdensity, assisting in the appropriate development o a clinicaldiagnostic. Expansion o the lesion was conrmed with areduction o nasopharynx space, observed in C images(Figure ).

Te imaging examinations also allow selecting the surgi-cal approach: intra- or extraoral. In our case we decided touse the intraoral approach, despite a large dimension cyst,which presented supercial involvement. During surgicalprocedures thecontents o the cysts revealed riableand whitematerial compatible with keratin (Figure ).


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(a) (b)

F : Microscopic ndings: (a) the lining o the cyst is composed o an epithelium which is attened and contains a granular layero keratohyalin granules, Haematoxylin and Eosin, x, and (b) oci o rupture and keratin exposed to the adjacent capsule and reactioncomposed o macrophages and oreign body giant cells (or keratin exposed), Haematoxylin and Eosin, x.

Te dermoid cyst can occur in the skin, ovary, testi-

cles, and other regions o the body always related to themidline, because it matches teratomatous injuries. In thecapsule, microscopic examination showed the presence oskin appendages in the cyst wall, in addition to the stratiedsquamous lining. Te gonadal lesions ofen exhibit othermature tissues such as cartilage, bone, atty tissue, andnerve tissue, as well as skin structure []. According to themicroscopic ndings, the present case is an epidermoid cystwith oci o rupture and keratin exposed to the adjacentcapsule with oreign body (keratin) and reaction composedo macrophages and oreign body giant cells (Figure ).

A total lesion excision was the recommended treatment,andthe recurrence is unusual. Te risk o malignancy o these

cysts is rare, but there are reports on dental literature. Teetiology and pathogenesis o the EC require more conclusivestudies [].

As mentioned, epidermoid cysts are benign lesions; how-ever, they may have large dimensions and cause physiologicalcomplications including swallowing and breathing difficulty.Te computed tomography is a method efficient or assessingthe relationship with the adjacent anatomical structures andplanning o surgical approach.

Conflict of Interests

Te authors have stated that they have no conict o interests.

Acknowledgments

Te Brazilian National Council or Scientic and echno-logical Development (CNPq) supported this project. CiroDantas Soares would like to thank CNPq or studentship/- Process.

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