the glandular odontogenic cyst: a case report

4
Oral Medicine The glandular odontogenic cyst: A case report Yasmiti Rodarte de Carvalho^ / Antonino Kimaid'^^^ / Luiz Antonio Guitnarâ Tere^inha de Oliveiia Nogueira* A case of a glandular odontogenic cyst is reported. All clinical, radiographie, and histopatho- logic features are di.scussed and compared with the descriptions in the literature already published about this new lesion. fQuiiitessence Itit 799'#;25,J5/-i Introduction The fiTSt and largest series of glandular odontogenic cysts (GOCs) was reported in the literature by Gardner et al^ in 1988, The authors considered this entity to be a rare cyst of the jaws that does not fit readily in the stan- dard classifications of odontogenic cysts. Some other cases have been reported in the literature,-^' but their tiimiber is not sufficient to establish definitive conclu- sions about all aspects of this new entity. As pointed out hy Gardner et al,' additional reports of this unusual cyst are necessary to complctncnt the information al- ready available about its biologic behavior. The term sialo-odontogenic cyst, used informally by Padayachce and Van Wick" is inappropriate, and shouldbeabandoned, according to Gardner et al,' The presence of mucous cells does not imply an origin from salivary glands, and Gardner et al' were emphatic that this new entity is undoubtedly odontogenic. The purpose of this article is to present a new case of GOC, discussing the clinical, radiographie, and histo- pathologic aspects of this entity, and compare the data with that found in the literature. * Professor, Departtnent of Patliolojîy, School of Dentistry of Sao Jose dos Campos, UNESP, Caixa Postal 314, Av. Fraticisco Longo, 777,12245 Sao José dos Campos, Sao Patilo, Brazil, ** Assistant Professor, Departtnent of Diagnosis and Surgery, School of Dentistry of Sao José dos Catrtpos, ** Professor, Department of Diagnosis and Surgery, School of Dentistry of Sao José dos Campos. Case report A 56-year-old woman was referred to the Oral Diagno- sis Clinic because of a painless swelling in the left man- dible. She repotted that the lesion had been present for approximately 1 year, fntraoral examination revealed an intraosseous growth with a buccal and lingual ex- pansion in the region of the mandibtilar left premolars and first molar, Exiraoral examination revealed asym- metry on the left side of the patient's face. Radiographie aspects Panoramic and occlusal radiographs showed a large, expansive, and well-defined multilocular radiolucent lesion (Figs 1 and 2), Histologie findings An excisional biopsy was performed and the tissue was prepared for light microscopy. The 5-\i.m paraffin sec- tions were stained with hematoxylin-eosin or periodic acid-Schiff (PAS) stain. Histologie analysis i,howed a cystic space lined by stratified squamous epithelium with few layers. In some areas epithehal cells were arranged in the form of ball-like structures nr swirhng areas (Fig 3, A and B). The superficial layer of the epithelium showed eosino- philic and cuboidal cells, and some of the spinous cells had vacuoli/ation in their cytoplasm ¡Fig 3, C). The epi- thelium slill showed abundant mucous cells (Fig 3, D and E). and sometimes mucous PAS-positive pools were lined by eosinophiiic cuboidal cells forming duct- like structures (Fig 3, F). The interface of the epithelia 351

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Page 1: The glandular odontogenic cyst: A case report

Oral Medicine

The glandular odontogenic cyst: A case reportYasmiti Rodarte de Carvalho^ / Antonino Kimaid' ^ / Luiz Antonio GuitnarâTere^inha de Oliveiia Nogueira*

A case of a glandular odontogenic cyst is reported. All clinical, radiographie, and histopatho-logic features are di.scussed and compared with the descriptions in the literature alreadypublished about this new lesion. fQuiiitessence Itit 799'#;25,J5/-i

Introduction

The fiTSt and largest series of glandular odontogeniccysts (GOCs) was reported in the literature by Gardneret al in 1988, The authors considered this entity to be arare cyst of the jaws that does not fit readily in the stan-dard classifications of odontogenic cysts. Some othercases have been reported in the literature,-^' but theirtiimiber is not sufficient to establish definitive conclu-sions about all aspects of this new entity. As pointed outhy Gardner et al,' additional reports of this unusualcyst are necessary to complctncnt the information al-ready available about its biologic behavior.

The term sialo-odontogenic cyst, used informally byPadayachce and Van Wick" is inappropriate, andshouldbeabandoned, according to Gardner et al,' Thepresence of mucous cells does not imply an origin fromsalivary glands, and Gardner et al' were emphatic thatthis new entity is undoubtedly odontogenic.

The purpose of this article is to present a new case ofGOC, discussing the clinical, radiographie, and histo-pathologic aspects of this entity, and compare the datawith that found in the literature.

* Professor, Departtnent of Patliolojîy, School of Dentistry ofSao Jose dos Campos, UNESP, Caixa Postal 314, Av. FraticiscoLongo, 777,12245 Sao José dos Campos, Sao Patilo, Brazil,

** Assistant Professor, Departtnent of Diagnosis and Surgery,School of Dentistry of Sao José dos Catrtpos,

** Professor, Department of Diagnosis and Surgery, School ofDentistry of Sao José dos Campos.

Case report

A 56-year-old woman was referred to the Oral Diagno-sis Clinic because of a painless swelling in the left man-dible. She repotted that the lesion had been present forapproximately 1 year, fntraoral examination revealedan intraosseous growth with a buccal and lingual ex-pansion in the region of the mandibtilar left premolarsand first molar, Exiraoral examination revealed asym-metry on the left side of the patient's face.

Radiographie aspects

Panoramic and occlusal radiographs showed a large,expansive, and well-defined multilocular radiolucentlesion (Figs 1 and 2),

Histologie findings

An excisional biopsy was performed and the tissue wasprepared for light microscopy. The 5-\i.m paraffin sec-tions were stained with hematoxylin-eosin or periodicacid-Schiff (PAS) stain.

Histologie analysis i,howed a cystic space lined bystratified squamous epithelium with few layers. Insome areas epithehal cells were arranged in the form ofball-like structures nr swirhng areas (Fig 3, A and B).The superficial layer of the epithelium showed eosino-philic and cuboidal cells, and some of the spinous cellshad vacuoli/ation in their cytoplasm ¡Fig 3, C). The epi-thelium slill showed abundant mucous cells (Fig 3, Dand E). and sometimes mucous PAS-positive poolswere lined by eosinophiiic cuboidal cells forming duct-like structures (Fig 3, F). The interface of the epithelia

351

Page 2: The glandular odontogenic cyst: A case report

Oral Medicine

Fig 1 Panoramic radiograph showing a radiclucent area inthe left mandible.

Fig 2 (right) Occlusal view ot the radiolucent lesion show-ing expansion of the vestibular corticai bone.

and [he subjacent connective fibrous tissue was flat andtherefore without rete pegs. This fibrous capsule wassparsely infiltrated by mononuclear inflammatoryceUs,

other entities. These general radiographie featureswere also observed in the present case.

According to Gardner et al,' the following histologiefindings should be detected during microscopic analy-

Foltow-up

After 1 year, the extraoral examination revealed asmall depression at the region of the previous surgery,A follow-up radiograph revealed bone formation in thesurgical area.

Discussion

The GOC described in this report fulfilled the diagnos-tic criteria proposed by Gardner et al,' From the lim-ited number of these cysts available for study in the lit-erature it seems that this lesion has a predilection formen,-"" However, in the present case, the patient was awoman. The mean age of occurrence is around 50 yearsold,"* but GOCs have been reported in patients rangingin age from the 20s to the 80s,-' The patient in this casewas in the sixth decade of life,

Radiographically, the lesion shows a unilocular ormultilocular pattern with well-defined limits, but doesnot show specific features thai allow distinction from

1, A lining epithelium of varying thicknesses exhibit-ing a flat interface with the underlying connectivetissue. Characteristically, there is no inflammatoryinfiltrate present within the connective tissue,

2, The presence of eosinophilic cuboidal ceUs in thesuperficial layer.

3, Pools of tiiucicarmine-positive material within theepithelium, often lined by eosinophihc cuhoidalcells similar to those found on the surface of the epi-thelium,

4, The presence of mucous eells, sometimes replacingthe eosinophilic cuboidal cells,

5, Basal cells that are sometimes hyperchromatic andmay be vacuolated. In some cases, cells of the spi-nous cell layer are also vacuolated,

6, in focal areas, epithelial cells that may be arrangedinto apparently spherical structures, called epithelialspheres-

1. Occasionally, irregularly shaped calcifications in theconnective tissue beneath the epithelium.

352 Quintessence International Volume 25, Number 5/1994

Page 3: The glandular odontogenic cyst: A case report

Oral Medicine

Fig 3 (A) and (ß) Squamous stratified epitheiium lining the glandular odcntogenic cyst witb focal thickening (hematoxyiin-eo-sin staining; original magnification x 275); (O) epithelial lining of the glandular odontcgenic cyst (hematoxylin-ecsin staining;original magnification x 275); (D) mucous PAS-positive cells in the lining of tbe giandular odontogenic eyst (periodic acid—Schiff staining; original magnification x 275); (£) mucous ceiis forming ductlike structures within the spinous cell layer (hema-toxylin-eosin staining; original magnification x 350); (F) the same area as shown in B (periodic acid - Schiff staining; originalmagnification x 350).

Fig 3a Fig 3b

Fig 3c Fig 3d

I»!*'

3E -' ^ "" ' • ^ ^ E \ ^ ' ' ^ '

Flg3eFig3f

?/SiOuintessepcalot jmber5/1994

353

Page 4: The glandular odontogenic cyst: A case report

Oral Medicine

In general, the cyst in the present report showed allthe above-mentioned histopathologic features, exceptthat the hyperchromatism and vacuolization of the ba-sal cells were absent and PAS-positive poois werefound. These differences arose because a different his-tocheniical stain was used.

The histogenesis of this new eyst is controversial anddebatable, but it has been emphatically suggested thatGOCs represent a rare type of odontogenic cyst. Thefindings of Gardner et al' lend support to an odonto-genic origin because ball-like epithelial structures arecharacteristic of odontogenic cysts and are found inother odontogenic lesions, such as lateral periodontalcysts^ and botryoid cysts,' Patron et al,'' in concordancewith Gardner et al,' suggested that GOCs are a variantolabotrvoidcvst.

References1, Gardner DG, Keffler HP, Morency R, Sdiaffmer BL, The glan-

dular odontogenic cyst: An apparent entity, J Oral Pathol

2, Ficarra G, Chou t., Pan7oni F.. Glandular odontogenic cyst (sia-lo-odontogenk cyst), A i ase report, Int J Oral Ma,\illolac Surgt990;t9:331-333,"

3, Souza SOM, Gayotto MV, Araujo VC, Araujo NS, Cistoodontogênico glandular. Relato de um caso e revisàoda literatti-ra. Rev Assoc Paul Cir Dent iyyü;44:27-2S,

4, Patron M, Colmenero C, Larrauri J. Glandular odontogenic cyst:Clinicopathological analysis of three cases. Oral Surg Oral MedOral Pathol li)Çll;72:71-74,

5, Padayachee A, Van Wick CW. Two eystic lesions with features otboth botryoid odontogenic cyst and the central mucoepidermoidtumour: Sialo-cdontogenic cyst, J Gral Pathol 1987;t6:499-504,

6, Weathers DR, Waldron CA. Unusual tnultilocular cysts of thejaws (botryoid odontogenic cysts). Oral Surg Orat Med OralPathol I973;36:235-241,

7, Wysocki OP, Brannon RB, Gardner DG, Fapp P, Histogenesis ofthe lateral periodontal cyst and the gingival cyst of the adult.Oral Surg Orai Med Oral Pathol 1980;50:327-334, D

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