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The peripheral odontogenic keratocyst Dan Dayan, Amos Buchner, Meir Gorsky and Mill HareI-Raviv Department of Oral Pathology and Oral Medicine, The Maurice and Gabriela Goldschleger School of Dental Medicine, Tel Aviv University, Tel Aviv, israel D. Dayan, A. Buchner, M. Gorsky and M. Harel-Raviv: The peripheral odontogenic keratoeyst. Int. J.-Oral Maxillofac. Surg. 1988; 17: 81-83. Abstract. A case with a firm asymptomatic nodule of 1 cm diameter on the gingiva between the left upper cuspid and first bicuspid is presented. Radiographic examination did not reveal any pathology of the bone in that region. Histologic examination revealed a cyst wall lined by squamous stratified epithelium, charac- teristic to the lining of an odontogenic keratocyst. It is suggested that the term peripheral odontogenic keratocyst be used for the diagnosis of this lesion. Key words: keratocyst; cyst, odontogenic; pri- mordial cyst Accepted for publication 10 August 1987 The WHO classification of jaw cysts de- fines a gingival cyst as one that orig- inates from the epithelial cell rests in the gingiva 14. Clinically, it appears as a small gingival swelling which occasion- ally results in some pressure resorption of the alveolar bone beneath the lesion. The epithelial lining may be of several types, the most common of which is a flattened lining with or without local- ized thickenings or buds, very similar to the lining of a lateral periodontal cyst 5,22. Additional types described include non- keratinizing stratified squamous epithe- lium and keratinized squamous epithe- lium s. 2 cases in which the lining is com- posed of parakeratinized epithelium with palisading basal cells, resembling the lining of an odontogenic keratocyst are reported by BUCHNER & HANSEN 5. The term odontogenic keratocyst was first used by PHILIPSEN 12, and subse- quently PINDBORG e t al.a5 established the histologic criteria: (1) the lining epithelium is usually very thin and uniform in thickness, with little or no evidence of rete ridges; (2) there is a well-defined basal-cell layer, the component cells of which are cuboidal or columnar in shape and of- ten in a palisaded arrangement; (3) there is a thin spinous-cell layer which often shows a direct transition from the basal-cell layer; (4) the cells of the spinous-cell layer frequently exhibit intracellular edema. (5) keratinization is predominantly parakeratotie, but it may be orthoker- atotic; (6) the keratin layer is often corru- gated; (7) the fibrous cyst wall is generally thin and usually not inflamed. This lesion has received special atten- tion in the literature because of a rela- tively high recurrence rate in compari- son to other types of odontogenic cysts 3, 4, 7. 8, 13, 16, 1~21. It has even been proposed that the odontogenic keratocyst be re- garded as a benign cystic neoplasm rather than a simple cyst ~,9. It is gener- ally accepted by most investigators that this cyst originates from odontogenic epithelium, namely residues or prolifer- ations of dental lamina ~7,~8. However, this has not been definitely established ~4. Our objective is to present a case of a gingival cyst of an adult, with an epi- thelial lining characteristic of an odon- togenic keratocyst and to suggest the diagnostic term of peripheral odonto- genic keratocyst. Case report A 42-year-old white male was referred to the Oral Medicine Clinic, School of Dental Medicine, Tel Aviv University, with an asymptomatic gingival nodule. The patient reported a 6-year duration of the gingival lesion. The lesion did not respond to anti- biotic therapy. The patient's medical history was non-contributory. Clinical examination revealed a 1 cm di- ameter nodule on the gingival area between the left upper cuspid and the first bicuspid (Fig. 1). The nodule was covered by a normal mucosa and was fluctuant to palpation. No other periodontal or dental pathology were noted in the area of the lesion, and the re- mainder of the oral mucosa was within nor- mal limits. Radiographic examination reveal- ed no bony lesions. An excisional biopsy was planned. A gin- gival flap was elevated, exposing a capsular soft tissue lesion located mainly on the buccal portion of the cuspid. Enucleation of the lesion revealed a small fenestration of the alveolar bone. Curettage of the area was per- formed before suturing the flap to its original location. Healing was within normal limits. Histologic examination of the enucleated tissue disclosed a soft tissue, consisting main- ly of a cyst wall, lined with stratified squa- mous epithelium (Fig. 2). The epithelium was of uniform thickness, approximately 6~10 cell layers and without fete ridges formation (Fig. 3). The basal-cell layer was well-defined and generally composed of cuboidal cells which exhibited polarization of nuclei. The spinous- cell layer was thin and frequently featured intracellular edema. The surface was covered by a parakeratotic-cell layer often corrugated or wrinkled. The connective tissue of the cyst wall was thin and fibrotic, generally free of inflammation, except in some areas where chronic inflammatory cells were present, even to the extent of destroying the covering epi- thelium. Because of its location in the gingiva and the histologic features of the cyst lining and wall, the lesion was diagnosed as a peri- pheral odontogenie keratocyst. No clinical recurrence was noted in a fol- low-up visit after 10 months. Fig. 1. Clinical photograph of a gingival nod- ule in the cuspid area.
Transcript

The peripheral odontogenic keratocyst

Dan Dayan, Amos Buchner, Meir Gorsky and Mill HareI-Raviv Department of Oral Pathology and Oral Medicine, The Maurice and Gabriela Goldschleger School of Dental Medicine, Tel Aviv University, Tel Aviv, israel

D. Dayan, A. Buchner, M. Gorsky and M. Harel-Raviv: The peripheral odontogenic keratoeyst. Int. J.-Oral Maxillofac. Surg. 1988; 17: 81-83.

Abstract. A case with a firm asymptomatic nodule of 1 cm diameter on the gingiva between the left upper cuspid and first bicuspid is presented. Radiographic examinat ion did not reveal any pathology of the bone in that region. Histologic examination revealed a cyst wall lined by squamous stratified epithelium, charac- teristic to the lining of an odontogenic keratocyst. It is suggested that the term peripheral odontogenic keratocyst be used for the diagnosis of this lesion.

Key words: keratocyst; cyst, odontogenic; pri- mordial cyst

Accepted for publication 10 August 1987

The W H O classification of jaw cysts de- fines a gingival cyst as one that orig- inates from the epithelial cell rests in the gingiva 14. Clinically, it appears as a small gingival swelling which occasion- ally results in some pressure resorption of the alveolar bone beneath the lesion. The epithelial lining may be of several types, the most common of which is a flattened lining with or without local- ized thickenings or buds, very similar to the lining of a lateral periodontal cyst 5,22. Addit ional types described include non- keratinizing stratified squamous epithe- l ium and keratinized squamous epithe- l ium s. 2 cases in which the lining is com- posed of parakeratinized epithelium with palisading basal cells, resembling the lining of an odontogenic keratocyst are reported by BUCHNER & HANSEN 5.

The term odontogenic keratocyst was first used by PHILIPSEN 12, and subse- quently PINDBORG e t al.a5 established the histologic criteria:

(1) the lining epithelium is usually very thin and uniform in thickness, with little or no evidence of rete ridges;

(2) there is a well-defined basal-cell layer, the component cells of which are cuboidal or columnar in shape and of- ten in a palisaded arrangement;

(3) there is a thin spinous-cell layer which often shows a direct t ransi t ion from the basal-cell layer;

(4) the cells of the spinous-cell layer frequently exhibit intracellular edema.

(5) keratinization is predominant ly parakeratotie, bu t it may be orthoker- atotic;

(6) the keratin layer is often corru- gated;

(7) the fibrous cyst wall is generally thin and usually no t inflamed.

This lesion has received special atten- t ion in the literature because of a rela- tively high recurrence rate in compari- son to other types of odontogenic cysts 3, 4, 7. 8, 13, 16, 1~21. It has even been proposed that the odontogenic keratocyst be re- garded as a benign cystic neoplasm rather than a simple cyst ~,9. It is gener- ally accepted by most investigators that this cyst originates from odontogenic epithelium, namely residues or prolifer- ations of dental lamina ~7,~8. However, this has not been definitely established ~4.

Our objective is to present a case of a gingival cyst o f an adult, with an epi- thelial l ining characteristic of an odon- togenic keratocyst and to suggest the diagnostic term of peripheral odonto- genic keratocyst.

Case report

A 42-year-old white male was referred to the Oral Medicine Clinic, School of Dental Medicine, Tel Aviv University, with an asymptomatic gingival nodule. The patient reported a 6-year duration of the gingival lesion. The lesion did not respond to anti- biotic therapy. The patient's medical history w a s non-contributory.

Clinical examination revealed a 1 cm di- ameter nodule on the gingival area between the left upper cuspid and the first bicuspid (Fig. 1). The nodule was covered by a normal mucosa and was fluctuant to palpation. No other periodontal or dental pathology were noted in the area of the lesion, and the re- mainder of the oral mucosa was within nor- mal limits. Radiographic examination reveal- ed no bony lesions.

An excisional biopsy was planned. A gin-

gival flap was elevated, exposing a capsular soft tissue lesion located mainly on the buccal portion of the cuspid. Enucleation of the lesion revealed a small fenestration of the alveolar bone. Curettage of the area was per- formed before suturing the flap to its original location. Healing was within normal limits.

Histologic examination of the enucleated tissue disclosed a soft tissue, consisting main- ly of a cyst wall, lined with stratified squa- mous epithelium (Fig. 2). The epithelium was of uniform thickness, approximately 6~10 cell layers and without fete ridges formation (Fig. 3). The basal-cell layer was well-defined and generally composed of cuboidal cells which exhibited polarization of nuclei. The spinous- cell layer was thin and frequently featured intracellular edema. The surface was covered by a parakeratotic-cell layer often corrugated or wrinkled. The connective tissue of the cyst wall was thin and fibrotic, generally free of inflammation, except in some areas where chronic inflammatory cells were present, even to the extent of destroying the covering epi- thelium. Because of its location in the gingiva and the histologic features of the cyst lining and wall, the lesion was diagnosed as a peri- pheral odontogenie keratocyst.

No clinical recurrence was noted in a fol- low-up visit after 10 months.

Fig. 1. Clinical photograph of a gingival nod- ule in the cuspid area.

82 Dayan, Buchner, Gorsky and Harel-Raviv

Fig. 2. Low power photomicrograph exhibit- ing a cyst wall lined with stratified squamous epithelium (H & E, x 40).

Discussion

Clinically, the present cyst fulfills the criteria of a gingival cyst of the adult: a soft tissue lesion which does no t pro- duce any radiographic changes. The size and durat ion probably resulted in ero- sion of the buccal aspect of the cuspid alveolar bone. Histologically, it showed the features of an odontogenic kerato- cyst.

According to SHEAR TM, the 2 cases re- ported by BUCHNER & HANSEN 5 in which the epithelial lining was similar to the epithelium of the odontogenic kerato- cyst should be diagnosed as a primor- dial cyst, despite having clinically, an apparent gingival cyst of the adult picture.

There are other peripheral odonto- genie cysts or tumors which are clin- ically located in the gingiva and diag-

nosed according to their histologic pictures: peripheral calcifying odonto- genic cyst (COC) TM, peripheral amelo- blastoma, peripheral calcifying epi- thelial odontogenic tumor (CEOT), peripheral adenomatoid odontogenic tumor (AOT) 6, peripheral dentinogenic ghost cell tumor 1°, and peripheral odon- togenic f ibroma (WHO-type). It is sug- gested that the term peripheral odonto- genie keratocyst be used for the diag- nosis of this lesion.

Many investigators point out that both the gingival cyst of the adult and the odontogenic keratocyst are formed from remnants of dental lamina ~7. The ability of these remnants to form keratin was observed by MALLASSEZ H. Further- more, keratinized epithelial islands de- rived from dental lamina are found (ex- traosseously as well as intraosseously) in jaws of fetuses 2. Further investiga- tions are suggested to elucidate whether the histogenesis of the peripheral odon- togenic keratocyst might be similar to the gingival cyst of the adult.

References

1. Ahlfors, E., Larsson, A. & Sjogren, S.: The odontogeuic keratocyst: A benign cystic tumor? J. Oral Maxillo-Fac. Surg. 1984: 42: 10.

2. Bhaskar, S. N.: Gingival cyst and the ker- atinizing ameloblastoma. Oral Surg., Oral Med., Oral Pathol. 1965: 19: 796.

3. Brannon, R. B.: The odontogenic kera-

F~g. 3. High power photomicrograph exhibiting the epethelial lining. Note mainly the corru- gated parakeratotic layer (H & E, x 40).

tocyst. A clinico-pathologic study of 312 cases. Part I. Clinical features. Oral Surg., Oral Med., Oral Pathol. 1976: 42: 54.

4. Browne, R. M.: The odontogenie kerato- cyst. Histological features and their cor- relation with clinical behaviour. Br. Dent. J. 1971: 131: 249.

5. Buehner, A. & Hansen, L. S.: The histo- morphologic spectrum of the gingival cyst in the adult. Oral Surg., Oral Med., Oral Pathol. 1979: 48: 532.

6. Buehner, A. & Sciubba, J.: Peripheral odontogenie tumors . - a review. Oral Surg., Oral Med., Oral Pathol. 1987: 63: 688.

7. Chuong, R., Donoff, R. B. & Guralnick, W. C.: The odontogenie keratoeyst. J. Oral Maxillo-Fac. Surg. 1982: 40: 797.

8. Donoff, R. B., Guralnick, W. C. & Clay- man, L.: Keratocysts of the jaws. J. Oral Surg. 1972: 30: 800.

9. Eversole, L. R. & Rovin, S.: Aggres- sive growth and neoplastic potential of odontogenic cysts with special re- ference to central epidermoid and muco- epidermoid carcinomas. Cancer 1975: 35: 270.

10. Hirshberg, A., Dayan, D. & Horowitz, I.: Peripheral dentinogenic ghost cell tumor. Int. J. Oral Maxillo-Fac. Surg. 1987: 16: 620.

11. Mallassez, L.: UExistence D'Amas Ep- itheliaux autour de la Racine Des Dents Chez UHomme Adulte eta l'etat Normal (Debris Epitheliaux Para Dentaires). Arch Physiol. (Normale et Pathologique) 1885: 3: (Serie 5~5): 129.

12. Philipsen, H. P.: Om keratoeyster (Kole- steatan) in Kaeberna. Tandlaegebladet 1956: 60: 963.

13. Pindborg, J. J. & Hansen, J.: Studies on odontogenic cyst epithelium. II. Clinical and roentgenologic aspects of odonto- genie keratoeysts. Aeta Pathol. Microbiol. Scand. 1963: 58: 283.

14. Pindborg, J. J., Kramer, I. R. H. & Tor- loni, H.: Histological typing of odonto- genie tumors, jaw cyst and allied lesion. WHO Geneva 1971: 40.

15. Pindborg, J. J., Philipsen, H. P. & Hen- riksen, J.: Studies on odontogenie cyst epithelium. I~.Keratinization in odonto- genie cysts. In: Butcher, E. O. & Sogness, R. F. (eds): Fundamentals of kera- tinization. American Association for the Advancement of Science, Washing- ton 1962, publication no. 70, pp. 151-160.

16. Rud, J. & Pindborg, J. J.: Odontogenic keratocysts: a follow-up study of 21 cases. J. Oral Surge. 1969: 27: 323.

17. Shafer, W. G., Hine, M. K. & Levy, B. M.: A textbook of oral pathology, 4th edition. WB Saunders Company, Phil- adelphia, 1983, p. 271.

18. Shear, M.: Cyst of the oral regions, 2rid edition. Wright PSG, Bristol, London, Boston 1983, p. 46.

Peripheral keratocyst 113

19. Shear, M.: Cyst of the jaws: Recent ad- vances. J. Oral Pathol. 1985: 14: 43.

20. Vedtofte, P. & Praetorius, F.: Recur- rence of the odontogenic keratocyst in relation to clinical and histological features. A 20-year follow-up study of 72 patients. Int. J. Oral Surg. 1979: 8: 412.

21. Voorsmit, R. A. C. A., Stoelinga, P. J.

W. & Van Haelst, V. J. G. M.: The man- agement of keratocysts. J. Maxillo-Fac. Surg. 1981: 9: 228.

22. Wysocki, G. P., Brannon, R. B., Gard- ner, D. G. & Sapp, P.: Histogenesis of the lateral periodontal cyst and the gingival cyst of the adult. Oral Surg., Oral Med, Oral PathoL 1980: 50: 327.

Address: Dr. Dan Dayan Department of Oral Pathology

and Oral Medicine The Maurice and Gabriela Goldschleger

School of Dental Medicine Tel Aviv University Ramat Aviv 69978 Tel Aviv Israel


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