Dentistry / Odontologia

34J Health Sci Inst. 2011;29(1):34-6

Synchronous pleomorphic adenoma and periapical cyst:

clinical case report

Adenoma pleomorfo e cisto periapical sincrônicos: relato de caso clínico

Cláudio Maranhão Pereira1, Danilo Santos Carneiro1, Patrícia Freire Gasparetto1, Tessa de Lucena Botelho1

1Dental School, University Paulista, Goiânia-GO, Brasil.

Abstract

The pleomorphic adenoma or mixed benign tumor is the most common benign neoplasia of the salivary gland in human beings. It preferen-tially occurs in the superficial lobe of the parotid gland. In the oral cavity, associated with the minor salivary glands, it has a greater predilec-tion for the palate region, with slight predisposition in women between the 3rd and 4th decades of life. The radicular cyst is an odontogenicinflammatory cyst resulting from pulp lesions caused by traumas or caries. In spite of being relatively common, there are no reports of associa-tion with salivary gland tumors. The aim of this work is to report a case of a 36-year-old woman patient, who presented with synchronous de-velopment of a pleomorphic adenoma and periapical cyst within the same region. The option taken was to perform enucleation of both lesions,followed by local curettage. The patient has been followed-up clinically and radiographically for around 18 months without signs of recurrence.

Descriptors: Adenoma, plesmorphic; Odontogenic cysts; Odontogenic tumours; Salivary gland, minor

Resumo

O adenoma pleomórfico ou tumor misto benigno é a neoplasia benigna mais comum primária de glândula salivar. Ocorre preferencialmente nolobo superficial da glândula parótida. Na cavidade oral, associado com as glândulas salivares menores, tem uma maior predileção para a regiãodo palato, com ligeira predisposição em mulheres entre a 3 ª e 4 ª décadas de vida. O cisto radicular é um cisto odontogênico inflamatório de-corrente de lesões causadas por traumas ou cárie. Apesar de ser relativamente comum, não há relatos de cistos periapicais associados com tu-mores de glândula salivar. O objetivo deste artigo é relatar um caso de uma paciente de 36 anos de idade com desenvolvimento sincrônico deadenoma pleomorfo e cisto periapical em uma mesma região. A opção tomada foi a de realizar a enucleação de ambas as lesões, seguido decuretagem local. A paciente está sendo acompanhada clínica e radiograficamente por cerca de 18 meses sem sinais de recidiva.

Descriptors: Adenoma pleomorfo; Cistos odontogênicos; Tumores odontogênicos; Glândulas salivares menores

Introduction

Salivary gland tumors constitute one of the most important groupsof lesions affecting the head and neck region. The pleomorphic ade-noma or mixed benign tumor is the most frequently occurring oneof the salivary gland tumors, affecting both the minor and major sa-livary glands. Among all the salivary glands, the parotid is the siteof major predilection1-4.

When it occurs in minor salivary glands, the hard palate region pre-sents the greatest predilection, followed by the upper lip, tongue, floorof the mouth and retromolar region. In the hard palate it may perforatethe bone and extend into the maxillary sinus1-2. Patients between theages of 30 and 40 years are the most affected, but it may develop at anyage, even in newborn children2. Clinically, these tumors present as so-litary oval lesions with well delimited margins. The lesion is mobile, ex-cept when it occurs in the palate, presenting slow growth and is asymp-tomatic. It may vary from a few to several centimeters in size1-3.

The recommended treatment protocol is surgical excision, re-currence being frequent and variable according to the location ofthe lesion and surgical technique used4-5. Fewer than 5% of thesetumors undergo malignant transformation, this being more commonin cases of multiple recurrences, and when this occurs it is deno-minated carcinoma ex pleomorphic adenoma5-6. There are reportsof simultaneous development of pleomorphic adenoma with sometypes of adenocarcinomas7-10. However few cases in the literaturedescribe associations or development of cysts in regions of gnathicbone4. The aim of this case report was to describe the case of a pa-tient with a pleomorphic adenoma associated with a periapical cystin the same region.

Case report

The patient, a 36-year-old woman, presented to the Oral Diag-nostic Service at the University Paulista, complaining of a “lump in

the mouth”. During anamnesis, no datum from the previous medi-cal and family history made any contribution.

On extraoral physical exam no asymmetry was found in the rightmaxillary region. The oral exam revealed the presence of an increasein volume in the shape of a cupola in the right maxillary posteriorpalatal region, covered with whole mucosa, with a firm consistencyon palpation, and measuring about 2.0 cm in its largest diameter.

After analyzing the imaging exam, a radiolucent, unilocular, cir-cumscribed, well defined lesion with a radiopaque halo was ob-served, associated with the roots of tooth 16, compatible with a pe-riapical cyst (Figure 1). Puncture and aspiration were performed, inwhich a yellow, viscous liquid was obtained. Immediately after-wards, an excisional biopsy of the lesion was performed, associa-ted with extraction of tooth 16. After cystic enucleation, it was pos-sible to visualize a mass with a firm consistency on palpation, welldelimited and in continuity with the extraction site (Figure 2).

In the trans-operative period it was necessary to increase the en-velope type incision to the maxillary tuber region, in addition to per-forming a relief incision with three angles to increase the surgicalexposure area, the maxillary sinus floor, major palatal artery as thenerve endings were preserved (Figure 3). After detachment of themucoperiosteal flap, the tumoral mass was visualized, in which enu-cleation and vigorous curettage was performed. Both lesions werefixed in 10% formol and sent to the Pathologic Anatomy Service.

The two tissues were microscopically analyzed, making it possi-ble to confirm the diagnosis of pleomorphic adenoma, in which theepithelial cells formed solid sheets or a double layer of ductiformstructures, containing eosinophilic inclusions within them. Thestroma of the lesion generally presented eosinophilic hyaline ma-terial (Figure 4). In the other tissue, the histologic characteristics con-firmed the diagnosis of periapical cyst, which was shown to be li-ned with stratified squamous epithelium associated with cholesterolcrystals with multinucleated giant cells, hemocytes and areas of he-mosiderin pigmentation, typical of the radicular cyst.

Figure 1. Radiographic aspect – a radiolucent, unilocular lesion wasobserved associated with the roots of tooth 16

Figure 2. The oral exam revealed the presence of an increase in vo-lume in the shape of a cupola in the right maxillary posteriorpalatal region

Figure 3. After cystic enucleation, it was possible to visualize a masswith a firm consistency on palpation, well delimited and incontinuity with the extraction site

Figure 4. It is possible to note the epithelial cells formed solid sheetsor a double layer of ductiform structures, containing eosi-nophilic inclusions within them

The post-operative period developed satisfactorily; the sutureswere removed after seven days, and fourteen days later the surgicalarea was found to be completely healed. The patient has been cli-nically and radiographically followed up for 18 months and no re-currence of the lesion has been observed.

Discussion

Both the pleomorphic adenoma and the periapical cyst present avery typical clinical pattern. Analyzing the pleomorphic adenoma, inthe case of palatal lesions, conventional radiographs are not of muchhelp in diagnosis, as the lesion is located in an area of many superim-positions, and in general, the palatal bone is not compromised3-4,8. Ne-vertheless, in this case, the option was taken to perform an orthopan-tomograph that proved there was no bone compromise.

The treatment most used for pleomorphic adenoma consists ofsurgical excision with a safety margin4-6,9, as recurrence could oc-cur due to remaining residues of the capsule or of the lesion itself.In the present case, conventional surgical removal was performedunder local anesthesia, due to the favorable characteristics the le-sion presented, since it is the most indicated therapy when the le-sion is situated in the minor salivary glands.

The determinant factor for recurrence is not the period of deve-lopment of the lesion in which the surgical treatment is performed,but the surgical technique used4-5,10. The prognosis is considered ex-cellent when the surgery is performed in an adequate manner, witha cure rate of approximately 95%2. It is of great value to follow upcases of pleomorphic adenoma, and post-operative control shouldcontinue for five years5.

Conclusion

In spite of these two intraoral pathologies being relatively com-mon when compared with the other cystic and glandular alterationsof the oral cavity, their development simultaneously and in the

Pereira CM, Carneiro DS, Gasparetto PF, Botelho TL. J Health Sci Inst. 2011;29(1):34-635

36 Synchronous pleomorphic adenoma and periapical cystJ Health Sci Inst. 2011;29(1):34-6

same region has not been reported in the specialized literature. Thus,the diagnosis of neoplastic and cystic lesions continues to play animportant role in the dental clinic. However, it is necessary to pointout the need for following up the patient, since the episodes of re-currence of some lesions occur at a late stage, generally in periodslonger than 5 years after treatment. Therefore, it is mandatory to haveknowledge of the clinical and histological characteristics and treat-ment of lesions involving the maxillae and their adjacent structures.

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9. Papadogeorgakis N, Kalfarentzos EF, Vourlakou C, Malta F, Exarhos D. Simul-taneous pleomorphic adenoma of the left parotid gland and adenoid cystic car-cinoma of the contralateral sublingual salivary gland: a case report. OralMaxillofac Surg. 2009;13(4):221-4.

10. Pires FR, Alves FA, Almeida OP, Lopes MA, Kowalski LP. Synchronous mu-coepidermoid carcinoma of tongue and pleomorphic adenoma of submandibu-lar gland. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2003;95(3):328-31.

Corresponding author:

Cláudio Maranhão PereiraDepartment of Oral Diagnosis

Dental Scool, University PaulistaSGAS Quadra 913, s/nº – Conjunto B – Asa Sul

Brasília-DF, CEP 70390-130Brazil

E-mail: odontologiabrasilia@unip.br; claudio.maranhao@go.estacio.br

Received November 24, 2010Accepted January 31, 2011