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Coll. Antropol.28 (2004) 1: 357362UCD 616.314-007.64

Original scientific paper

Calcifying Odontogenic Cyst Gorlin's Cyst Report of Two Cases

Goran Kne`evi}1, Klara Sokler1, Pavel Kobler2 and Spomenka Manojlovi}3

1 Department of Oral Surgery, University Hospital Dubrava, Zagreb, Croatia2 Department of Oral Surgery, School of Dental Medicine, University of Zagreb, Zagreb, Croatia3 Department of Pathology, School of Medicine, University of Zagreb, Zagreb, Croatia

A B S T R A C T

The authors present two cases of calcifying odontogenic cysts, which were confirmedby histological examination. In the first case the radiographic findings and clinical sta-tus did not indicate the presence of a calcifying odontogenic cyst. In the second case,differential diagnosis included COC. The histopathological findings showed that whatappeared to be simple cases of bone translucencies was in fact an unusual odontogeniclesion. The two cases point out the possibility of incorrect assessment of deceptively ba-nal cases in daily specialist practice.

Key words:calcifying odontogenic cyst, Gorlin's cyst, keratinizing epithelial odon-togenic cyst, calcifying ghost cell odontogenic tumor, epithelial odontogenic ghost celltumor

Introduction

A calcifying odontogenic cyst (COC) israrely found in everyday practice in oralsurgery. It was thoroughly described by

Gorlin and coworkers in 19621 and in1963 Gold introduced the term kerati-nizing and calcifying odontogenic cyst2.He believed that this specific type of cysthad not been described previously in theliterature as an entity. The World HealthOrganization (WHO) according to Kra-mer and Pindborg classification from19923 and the majority of the authors fa-

vored the use of the term calcifying odon-togenic cyst322 and described it as a cys-tic or neoplastic-like odontogenic patho-

logical lesion of the jaw and classified itas a benign odontogenic tumor3.

Calcifying odontogenic cyst represents2% of all odontogenic pathological chan-ges of the jaws4, although it can be foundtogether with other odontogenic tumors,most frequently with odontoma in 24% ofcases5.

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Received for publication October 22, 2002

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It most often occurs as a central (in-traosseous) lesion3,4,6,7,9, whereas periph-eral (extraosseous) localization in the soft

tissue is rare

8,14

, their incidence in the to-tal number is 1325%3,7,10,9. In 65% ofcases calcifying odontogenic cyst occursin the anterior part of the jaw in the inci-sor and canine region5,6,7,13. Central andperipheral forms of calcifying odontoge-nic cyst occur equally in the upper andlower jaws4,8,10,12. Johnson14 reported theoccurrence of 60% in the mandible, 30%in the form of peripheral calcifying odon-togenic cysts, while the anterior part of

the jaw was involved in 53% of cases. Onthe basis of 52 examples of calcifyingodontogenic cysts connected with anodontoma Hirshberg et al.15 concludedthat the upper jaw was affected in 61.5%and the anterior region of the jaw in 75%.

COC cyst can occur in very young pa-tients, even in the first year of life17.Cases have also been recorded of patientsin their eighties5,7. However, in the ma-

jority of cases it occurs in the second de-

cade of life5,7,17,18.

COC may clinically be diagnosed ascalcifying epithelial odontogenic tumor11,adenomatoid odontogenic tumor, ameloblas-tic fibroodontoma, complex or compoundodontoma, dentigerous cyst or as othertypes of odontogenic cysts. It is unclearwhether it should be regarded as a sepa-rate entity or as a stage in development ofanother odontogenic tumor1113,2325.

The reasons why in the past we didnot have the diagnosis of COC were prob-ably in clinical and histological similarityof the lesion with some odontogenic tu-mors or cysts. The lesion differs histo-logically from the odontogenic cysts andepithelial tumors as ameloblastoma, butcould be similar to the calcifying epithe-lial odontogenic tumor that presentsmore aggressive growth. Presentation offollowing two cases diagnosed in last twoyears could be our contribution for better

understanding of this lesion in our clini-cal practice.

ResultsCase 1

A 49-year-old man was referred to theDepartment of Oral Surgery for a swell-ing in the vestibulum of the left side ofthe mandible. No visible facial altera-tions could be seen, but a mass of theouter wall of the mandible was detectedby palpation.

Intraorally a sharply circumscribed

swelling could be seen on the left side ofthe mandible, 6.8 2.5 cm. The swellingwas covered with unchanged mucosa.

The lesion stretched from the left lat-eral incisor to the lower third molar onthe same side of the jaw. Both premolarsand the first and second molar of the jawwere missing. The loosening of the corti-cal bone could be felt by palpation. The

jaw appeared distended. The left canine,the root of which protruded into the cyst,did not react to electrical stimulus, al-though the tooth was not movable. Noparesthesia of the lower lip was present.

The radiographic findings showedsharply circumscribed bilocular translu-cency along the lower edge of the mandi-ble. The root of the left canine was notresorbed (Figure 1).

Differential clinical diagnosis includedan odontogenic residual cyst, odontogenic

keratocyst, monocystic ameloblastoma12or some other unusual soft odontogenictumor3,7,12,21,26,27.

Because of the size of the cyst the op-eration was performed under general an-esthesia. The cystic lesion was enuclea-ted and a permanent postoperative drainby plastic tube was used28.

The material was a hollow formation4.2 cm in diameter with brown content inthe lumen. Histologically the wall wascomposed of fibrous tissue, and the inter-

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nal surface covered in places by a thinlayer or in others by a thick layer of epi-thelial cells. There were hilic, and in pla-ces calcified areas, and ghost cells26. Tinyislets of odontogenic epithelia could beseen in the connective stroma (Figure 2).

Case 2

A 21 year-old girl was referred to theDepartment of Oral Surgery after an X-

ray examination showing a cyst of the up-per jaw.

Clinically, the deformations could be

seen on the vestibular wall of the upperjaw in the region of the right impacted ca-nine extending to the right first molar.The patient was diabetic. Sharply cir-cumscribed translucency could be seen onthe X-ray bite image, which stretchedfrom the region of the right impacted ca-nine (the deciduous canine was also pres-

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Fig. 1. Calcifying odontogenic cyst case 1.Orthopantomogram shows bilocular translu-

cency in the left body of the mandible.

Fig. 2. Calcifying odontogenic cyst case 1.The cystic formation, covered by normal epi-

thelium, the basal layer of which is composedof tiny cubic cells with dark nuclei, and sur-

face layers of large, bright, loosely distributedcells, similar to a star-like reticulum. On thesurface are accumulations of large eosinophi-lic masses containing keratinized shadow

cells (HE 20).

Fig. 3. Calcifying odontogenic cyst case 2.The bite image of the maxilla shows translu-cency in the bone at vestibular swelling with

a thinned cortical bone.

Fig. 4. Calcifying odontogenic cyst case 2.Translucency visible on the intraoral image

in the premolar and canine area. The shadowof the crown of the impacted canine and sha-dow of calcified dental tissue under the crown

of the canine can be seen on the edge of thetranslucency.

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ent) up to the region of the first right mo-lar (Figures 3 and 4).

No fluctuation of a cystic content was

noticed. Loosening of the cortical buccalwall was observed as well as swelling ofthe palate.

Differential clinical diagnosis inclu-ded follicular cyst and odontoma of theupper jaw12, with the possibility of one ofthe odontogenic tumors in which calcifieddental tissue occurs11,12,26.

The operation was performed undergeneral anaesthesia by enucleation of thelesion, in agreement with the principle ofclinical method for treating small cysticlesions of jaws28.

The material was an oval irregularlydeveloped firm mass, resembling a tooth,and a long hollow formation 2.5 cm in di-ameter. Histologically the lesion was con-sisted of cellular fibrous tissue covered byodontogenic epithelium which in placesstrands composed of ghost cells and foci ofcalcification. In places islets of odonto-

genic epithelium could be seen in the con-nective stroma (Figures 5 and 6).

Discussion

We have described here first two casesof calcifying odontogenic cysts found atclinical material of the Department ofOral Surgery.

In the first case, although the clinicalimage indicated an odontogenic residualcyst, odontogenic keratocyst or monocys-tic ameloblastoma, a calcifying odontoge-nic cyst was found. In the second case thepossibility of a calcifying odontogenic cystwas suspected and histopathologicallyconfirmed. The specificity of these twocases is that the clinical diagnosis shownin the radiographs appeared to be rela-tively simple and clear, particularly inthe first case. However, the histopatholo-gical diagnosis proved to be a rare odon-togenic lesion.

The presented cases are examples oftwo types of calcifying odontogenic cyst(monocystic and monocystic odontomacreative).

In 1981 Praetorius and coworkers6 triedto classify calcifying odontogenic cyst bydividing it into two entities: a cyst and aneoplasm. The cystic entity was classifiedinto three types. Type 1: a simple mo-nocystic type of typical Gorlin's cyst, withor without dentinoid calcified tissue, Ty-pe 2: monocystic odontoma creative type,

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Fig. 5. Calcifying odontogenic cyst case 2.The lumen of the cyst is covered by cubical

and cylindrical odontogenic epithelium withaccumulations of keratinized and calcified

large eosinophilic cells. (HE 100).

Fig. 6. Calcifying odontogenic cyst case 2.Eosinophilic masses composed of accumula-

tions of keratin and rests of ghost cells(HE 250).

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with all the characteristics of the previ-ous type, except that the hard tissue wascomplex or compound odontoma, and a

presence of ameloblastic fibroma tissue inthe cystic wall extending into the sur-rounding tissue, Type 3: monocystic ame-loblastomatous proliferating type whichwas marked by ameloblastomatous pro-liferation both in the walls and in the lu-men, and hard dental tissue which con-sisted of dentinoid formation in connec-tion with islets of epithelia in the connec-tive wall. The neoplastic form was de-scribed as an odontogenic tumor with so-

called shadow-like cells ghost cells. Theepithelial elements consisted of numer-ous ameloblastomatous proliferations oftissue in the connective tissue of the stro-ma. Within the epithelial islets were pre-sent different amounts of ghost cells, andthe hard tissue was composed of differentamounts of dentinoid in direct contactwith the epithelium6,12.

According to Pretorius6 our first casecould be classified as Type 1 and the sec-

ond as Type 2.

In 1992 Bucher5 described a multi-cystic form of calcifying odontogenic cystas a separate entity, and on the basis ofan analysis of 215 clinical and histologi-cal features of central calcified odontoge-nic cysts5. Instead of calcifying odontoge-nic cyst Langlais and coworkers suggestedthe term calcifying odontogenic lesion in-cluding cystic and tumorus form as sepa-

rate forms, and a combined lesion whenelements of both forms are observed12.

Calcifying odontogenic cyst is mostfrequently radiographically seen as a uni-locular translucency5,17,19 with sharplycircumscribed edges7,17 occurring in the

form of a multilocular lesion in a verysmall number of cases, from 5% to13%18.

A definite diagnosis of calcifying odon-

togenic cyst can be reliably made on thebasis of a histological examination.

With regard to the very small numberof recurrences, only eight cases of recur-rences have been documented in Englishliterature1,22,23,24.

Treatment of calcifying odontogeniccyst is usually enucleation of the cyst. Inhis most recent publication on pathologyBarnes mentions the possibility of malig-

nant transformation, and in differentialdiagnosis points out that COC must bedifferentiated from calcifying odontoge-nic tumor and ameloblastoma, that areessentially more aggressive and requirean extensive surgical approach26.

The diagnosis of pathological altera-tions of odontogenic etiology appears verysimple. However, only to those with a su-perficial knowledge of the matter. Thefurther one considers the problem of odon-

togenic tumors, so an excellent knowl-edge of the structure is necessary for cor-rect diagnosis, both on the part of the cli-nician and the pathologist, who need tohave subspecialist training in this field.

An optimal solution would be the exis-tence of a clinical pathologist, who wouldarrive at the final diagnosis in coopera-tion with an oral or maxillofacial sur-geon.

COC may mimic numerous odontoge-nic or not lesions therefore the clinicaland histological diagnosis is difficult.These two cases demonstrate in additionthat a specific knowledge in oral histopa-thology is required to differentiate odon-togenic lesions.

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R E F E R E N C E S

1. GORLIN, R. J., J. J. PINDBORG, F. P. CLAU-SEN, R. A. VICKERS, Oral Surg. Oral Med. Oral Pa-

thol., 15 (1962) 1235. 2. GOLD, L., Oral Surg. OralMed. Oral Pathol., 16 (1963) 1414. 3. KRAMER, I.R. H, J. J. PINDBORG, M. SHEAR: Histological typ-ing of odontogenic tumors. (Springer Verlag, Berlin1992). 4. ALTINI, M., A. G. FARMAN, Oral Surg.Oral Med. Oral Pathol., 40 (1975) 751. 5. BU-CHER, A., J. Oral Maxillofac. Surg., 49 (1991) 330. 6. PRAETORIUS, F., E. HJORTING-HANSEN, R. J.GORLIN, R. A. VICKERS, Acta Odontol. Scand., 39(1981) 227. 7. FREEDMAN, P. D., H. LUMERMM,J. K. GEE, Oral Surg. Oral Med. Oral Pathol., 40(1975) 93. 8. SWAN, R. H., G. D. HOUSTON, S. P.MOORE, J. Periodontol., 56 (1975) 340. 9. ERAS-MUS, J. H., I. O. C. THOMPSON, L. J. VAN RESEN-

BERG, A. J. VAN DER WESTHUIJZEN, Dentoma-xillofac. Radiol., 27 (1998) 30. 10. NEVILLE, B. W.,D. D. DAMM, C. M. ALLEN, J. E. BOUQUOT: Oraland maxillofacial pathology. (W. B. Saunders, Phila-delphia, 1995). 11. PINDBORG, J. J., Cancer, 11(1958) 838. 12. LANGLAIS, R. P., O. E. LANG-LAND, C. J. NORTJE: Diagnostic imaging of theJaw. (Williams & Wilkens: Malvern, 1995). 13.HONG, S. P., G. L. ELLIS, K. S. HARTMAN, OralSurg. Oral Med. Oral Pathol., 72 (1991) 56. 14. BU-CHER, A., P. W. MERRELL, L. S. HANSEN, A. S.LEIDER, Oral Surg. Oral Med. Oral Pathol., 72

(1991) 65. 15. JOHNSON, A., M. FLETCHER, L.GOLD, S.Y. CHEN, J. Oral Maxillofac. Surg., 55

(1997) 679. 16. HIRSHBERG, A., L. KAPLAN, A.BUCHER, J. Oral Maxiflofac. Surg., 52 (1994) 555. 17. LELLO, G. E., M. MAKEK, Int. J. Oral Maxillo-fac. Surg., 15 (1986) 637. 18. NAGAO, T., T. NAKA-JIMA, M. FUKUSHIMA, T. ISHIKI, J. Maxillofac.Surg, 11 (1983) 174. 19. TANIMOTO, K., S. TOMI-TA, M. AOYAMA, Y. FURUKI, M. FUJITA, T. WADA,Int. J. Oral Maxilofac. Surg., 17 (1988) 29. 20.DOMINGUEZ, F. V., E. G. ESPINAL, Acta Odontol.Latinoam., 1 (1984) 77. 21. McGOWAN, R. H., R.M. BROWNE, Br J. Oral Surg., 20 (1982) 203. 22.SLOOTWEG, P. J., R. KOOLE, J. Maxillofac. Surg., 8(1980) 143. 23. STOELINGA, P. J., F. B. BRONK-HORST, J. Craniomaxfac. Surg., 16 (1988) 184. 24.

SWIMSON, T. W., Br J. Oral Surg., 13 (1976) 217. 25. HOFFMAN, S., J. R. JACOWAY, S. O. KROGS:Intraosseous and parosteal tumors of the jaws.(ArMed. Forces Institute of Pathology, Washington,1987). 26. BARNES, L.: Surgical pathology of thehead and neck. (Mareel Dekker Inc, New York-Basel,2001). 27. RUSHTON, E., K. HORNER, Br. J. OralMaxillofac. Surg., 35 (1997) 196. 28. GRUREVI],J., G. KNE@EVI]: Alternative u lije~enju velikih ci-sta donje ~eljusti. In: Proceedings. (9. kongres Udru-ènja stomatologa Jugoslavije, Ljubljana, 1988).

G. Kneèvi}

Department of Oral Surgery, University Hospital Dubrava, Av. G. [u{ka 6,10040 Zagreb, Croatia

e-mail: [email protected]

KALCIFICIRAJU]A ODONTOGENA CISTA GORLINOVA CISTA PRIKAZ DVAJU SLUÂJEVA

S A @ E T A K

Autori prikazuju dva slu~aja kalcificiraju}ih odontogenih cista koje su potvr|enehistolo{kom ra{~lambom. U prvom slu~aju rentgenski nalaz i klini~ka slika nisu nago-vje{tavali postojanje kalcificiraju}e odontogene ciste. U drugom slu~aju diferencijalnadijagnoza uklju~ivala je i mogu}nost kalcificiraju}e odontogene ciste. Histopatolo{kinalazi pokazali su, da su ono {to se ~inilo da su bili jednostavni primjeri ko{tanih pro-svjetljenja zapravo bile neobi~ne odontogene patolo{ke promjene. Prikazana dva slu-~aja isti~u mogu}nost neto~ne procjene prividno banalnih slu~ajeva u svakodnevnojspecijalisti~koj praksi.

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