gorlin´s cisty

Calcifying odontogenic cyst A review of ninety-two cases with reevaluation of their nature as cysts or neoplasms, the nature of ghost cells, and subclasslfication

Sam Pyo Hong, DDS,a Gary L. Ellis, DDXb and Kenton S. Hartman, COL, USAF, DC,’ Washington, D.C.

ARMED FORCES INSTITUTE OF PATHOLOGY

Ninety-two cases of calcifying odontogenic cyst (COC) were reviewed with special consideration of their nature as cysts or neoplasma, the nature of ghost cells, and classification on the basis of clinicopathologic features. The cases were divided into 79 (85.9%) cysts and 13 (14.1%) neoplasma. The cysts occurred as four variants: (1) nonproliferative COC (35 cases), characterized by a simple unicyatic structure; (2) proliferative COC (17 cases), characterized by a cystic structure with multiple daughter cysts, extensive ghost cell formations, and marked tendency for calcification; (3) ameloblaatomatoua COC (11 cases), characterized by ameloblaatoma-like, cyst-lining epithelium with ghost cells and calcifications; and (4) COC associated with odontoma (16 cases), which combined features of COC and odontoma. The neoplasma occurred as three variants: (1) ameloblaatoma ex COC (two cases), which showed unifocal and multifocal intraluminal and intramural ameloblaatoma proliferating from the COGlining epithelium; (2) peripheral epithelial odontogenic ghost cell tumor (eight cases), which occurred in the gingiva and resembled peripheral ameloblaatoma except for clustered ghost cells in the central portion of epithelial islands and the presence of juxtaepithelial dentinoid; and (3) central epithelial odontogenic ghost cell tumor (three cases). The latter showed ameloblaatomatoua or adenomatoid odontogenic tumor-like epithelial clusters with ghost cell formation and juxtaepithelial dentinoid. The clinical features of cystic and neoplaatic variants were tabulated and described. On the basis of hiatopathologic features and their immunohiatochemical reaction to polyclonal antikeratin antibody, it is suggested that ghost cells might be the result of coagulative necrosis. (ORAL SURC ORAL MED ORAL PATHOL 1991;72:56-64)

C onsiderable histopathologic diversity has been recognized among lesions referred to as calcifying odontogenic cyst (COC) or under related diagnostic terms. Terms that have been used to designate these lesions include variants of cholesteatoma,’ mixed odontogenic tumor,2 atypical adamantinoma,3 calcifying odontogenic cyst, 4, 5 keratinizing and calcifying odontogenic cyst,(j keratinizing ameloblastoma,’ calcifying ghost cell odontogenic tumor,8 and cystic calcifying odontogenic tumor.9 The existence of numer-

The opinions or assertions contained herein are the private views of the authors and are not to be construed as official or as reflecting the views of the Departments of the Army, Air Force, Defense, or Veterans Affairs. aFormerly Visiting Fellow, Department of Oral Pathology; now at the Department of Oral Pathology, College of Dentistry, Seoul National University, Seoul, Korea. bDepartment of Oral Pathology and Department of Veterans Af- fairs, Special Reference Laboratory for Pathology. cAssociate Director. 7/14/26714

56

ous terms reflects a diverse histopathology and confu- sion about their nature as cysts, neoplasms, or hamartomas. Although most pathologists favor the term calcifying odontogenic cyst, which emphasizes the cystic nature, odontogenic origin, and calcifying potential of these lesions, others have interpreted them as neoplasms. 8* lo-l2 Some COCs have been reported to occur in close association with other odontogenic tumors, such as odontomas4, 5, 8, 9* I33 I4 and ameloblastic fibro-odontoma,lOy I5 and to recur after surgical intervention.3‘5, ‘6 ” Even malignant transformation of COC has been documentedI and mentioned. l9

The question concerning the nature of COCs seemed to be solved by Praetorius et al.,” who classified these lesions into two different entities: cysts and neoplasms. However, the classification and description was found to be somewhat ambiguous and unsuitable for classifying many of the cases of COC in the files of the Armed Forces Institute of Pathol- ogy (AFIP). For example, Praetorius et al. classified COCs with prominent intramural and intraluminal

Volume 72 Number 1

Calcifying odontogenic cyst 57

Table I. Subtypes and number of cases of so-called cot

No. (%,I of cases

I. Cystic A. Nonproliferative B. Proliferative C. Ameloblastomatous D. Associated with odontoma

II. Neoplastic A. Ameloblastoma ex COC B. Peripheral epithelial odontogenic

ghost cell tumor C. Central epithelial odontogenic

ghost cell tumor Total

79 (100.0) 35 (44.3) 17 (21.5) 11 (13.9) 16 (20.3) 13 (100.0) 2 (15.4) 8 (61.5)

3 (23.1)

92

ameloblastomatous proliferation as cysts. However, it appears that some cases of COC with prominent ameloblastomatous proliferation might be neoplastic. Therefore we have proposed what we consider to be a more reproducible classification of so-called COC.

In this study a large number of so-called COCs from the files of the AFIP were reviewed for the pur- poses of formulating a more reproducible classification system, assessing the pathogenesis of ghost cells, and reevaluating the microscopic spectrum and clinical parameters.

MATERIAL AND METHODS

One hundred twenty cases diagnosed as COC or under related diagnostic terms were retrieved from the files of the AFIP Registry of Oral Pathology and reexamined. These were cases submitted to the AFIP by civilian and military pathologists for consultation. Twenty-eight cases lacked adequate clinical information, or the microscopic slides were of poor qual- ity, and were excluded from the study. Ninety-two cases were considered suitable for this study. Stan- dard hematoxylin-and-eosin-stained tissue sections were available for review in all cases. Peroxidase-antiperoxidase immunohistochemical studies with polyclonal antikeratin antibody were performed on eight selected cases to examine the antigenicity of the ghost cells.

Fig. 2. High-magnification view of epithelial lining of nonproliferative COC shows odontogenic epithelium with scattered ghost cells and calcified bodies. (Hematoxylin- eosin stain; original magnification, X300.)

Cystic variants

RESULTS

Of the 79 cystic lesions in this series, 35 (44%) were characterized by a simple unicystic structure lined by odontogenic epithelium of 4 to 10 cells thick that contained isolated or clustered ghost cells and dystrophic-type calcification (Figs. 1 and 2). Juxtaepithelial dentinoid and foreign body reactions were seldom present, but this type of COC frequently occurred with cholesterol granulomas or hemorrhage. They were classified as nonproliferative COC.

The cases could be divided into two basic groups, Seventeen (22%) cystic lesions were distinctive be- cysts and neoplasms, on the basis of clinicopathologic cause of their proliferative cyst-lining epithelium and features. The cystic lesions could be separated further the presence of multiple daughter cysts of variable into four subgroups on the basis of their proliferative size in the fibrous connective tissue wall (Fig. 3, A). activity and growth pattern of the cyst-lining epithe- In the centers of the cysts were usually extensive ghost lium. The neoplasms were divided into three sub- cell formations with a marked tendency for calcifica- groups on the basis of site (intraosseous or ex- tion. The calcification of ghost cells appeared dystro- traosseous) and histologic features (Table I). phic and was more prominent than in other types of

Fig. 1. Nonproliferative COC is characterized by simple unicystic structure with relatively thin epithelial lining. These cysts occur both intraosseously (A) and extraosseously in gingiva or alveolar ridge (B). (Hematoxylin-eosin stain; original magnification, X 1.5.)

58 Hong, Ellis, and Hartman ORAL SURC ORAL MED ORAL PATHOL July 1991

Fig. 3. A, Proliferative COC demonstrates relatively thick epithelial lining with extensive ghost cell formation, hyaline material, and daughter cysts (center and right). B, Dystrophic-type calcification of ghost cells. (Hematoxylin- eosin stain; original magnifications: A, X30; B, X150.)

COC (Fig. 3, I?). Juxtaepithelial dentinoid was seldom found. Multiple ghost cell islands often formed solid tumor masses in the connective tissue wall. Foreign body reactions to herniated ghost cells were more prominent in this variant than in other types of COC. Atrophy of basal cells of the cyst-lining epithelium was also more extensive than in other types. This group of COCs was classified as proliferative type.

A third group of 11 (14%) cystic lesions was characterized by a unicystic structure in which the lining epithelium showed unifocal or multifocal intraluminal proliferative activity that resembled ameloblastoma but that also contained isolated or clustered ghost cells and calcification (Fig. 4). The tendency of ghost cells to calcify was not as great as in the proliferative COC. Juxtaepithelial dentinoid was occasion- ally found, and foreign body granulomas to herniated ghost cells were not prominent. Although the basal cells showed ameloblastic proliferative activity, they

Fig. 4. Cystic COC with ameloblastomatous appearance of proliferating odontogenic epithelium has plexiform pattern with areas of ghost cell formation and calcified bodies. Basal cell hyperchromatism, vacuolization, and nuclear polarization, which are often seen in ameloblastoma, are ab- sent here. (Hematoxylin-eosin stain; original magnification, X75.)

Fig. 5. Rudimentary tooth structure (center) is associated with nonproliferative COC. (Hematoxylin-eosin stain; original magnification, X40.)

did not completely satisfy the histopathologic criteria of early ameloblastoma as suggested by Vickers and Gorlin.20 This group was designated ameloblastomatous cot.

The remaining 16 (20%) cystic lesions were characterized by the combined microscopic features of

Volume 72 Number I

Number of Patients


-1st 2nd 3rd 4th 5th 6th 7th 8th 9th

Decade

Type IA Type IB W Type IC n Type ID

Fig. 6. Line graph of ages of patients with cystic COC shows broad age distribution with predominance in second decade of life. COC associated with odontoma (type ID) occurs only in first three decades of life. Type IA, Nonpro- liferative COC; Type IB, proliferative COC; Type IC, ameloblastomatous COC; Type ID, COC with odontoma.

Fig. 8. Epithelial proliferation with features of plexiform ameloblastoma extend into connective tissue wall (A) from simple COC of mandible (B). (Hematoxylin-eosin stain; original magnification, X75.)

Fig. 7. Radiograph shows well-circumscribed radiolu- cency with central radiopaque mass and inferior impacted canine tooth in anterior mandible that illustrates COC associated with odontoma.

COC and odontoma (Fig. 5). Most of the COCs in this group were simple unicystic structures. The feature that distinguished this lesion from an odontoma with ghost cells was the definite formation of a cyst.

At the time of diagnosis the age was known for 76 of the 79 patients. The cystic variants occurred in all age groups, and patients’ ages ranged from 2 days to 84 years (Fig. 6). The mean age in all cystic variants was 33 years. The mean age for patients with COC associated with odontoma was 14.7 years, which was significantly younger than that with the other types.

The sex was known for 78 patients. Forty-seven (60%) and 3 1 (40%) lesions occurred in males and fe- males, respectively, with no significant difference among the histologic types.

Sixty-nine (87%) cysts occurred intraosseously: 30 in the maxilla, 37 in the mandible, and 2 in which

mandible or maxilla was not specified. Ten (13%) lesions were located extraosseously in the gingiva and were distributed evenly between maxilla and mandible. Of the extraosseous COCs, eight were nonproliferative (Fig. 1, B) and two were proliferative. There was no significant preference for either maxilla or mandible among the cystic COCs except in the ameloblastomatous type, in which 9 of 11 lesions occurred in the mandible. Any preference for the anterior or posterior regions of the jaws could not be de- termined from the available information.

All cystic COCs occurred as circumscribed, expansile intraosseous lesions or tender gingival swellings. Most were asymptomatic. One intraosseous lesion caused spontaneous exfoliation of an affected tooth, whereas another cyst was associated with mobility of an affected tooth. One intraosseous lesion recurred 10 years after excision. All extraosseous lesions occurred without bone erosion.

Radiographically, intraosseous lesions appeared as well-circumscribed radiolucencies with or without radiopaque masses or malformed teeth (Fig. 7).

60 Hong, Ellis, and Hartman ORAL SURC ORAL MED ORAL PATHOL July 1991

Table II. Clinical features of peripheral epithelial odontogenic ghost cell tumors

Age No. (p-J/sex Location Clinical manifestation

Fig. 9. A, Peripheral epithelial odontogenic ghost cell tumor is characterized by epithelial islands and proliferations from surface gingival epithelium into lamina propria. B, Ameloblastic epithelial clusters contain ghost cells and are associated with dentinoid calcifications. (Hematoxylin- eosin stain; original magnifications: A, X30; B, X75.)

Neoplastic variants

Of the 13 lesions identified as neoplasms, two were classified as ameloblastoma arising in COC (Fig. 8). The cyst-lining epithelium of these lesions showed unifocal and multifocal intramural and intraluminal proliferation, usually in a plexiform pattern, with histopathologic features of early ameloblastoma as suggested by Vickers and Gorlin.*O These features in- cluded hyperchromatism, palisading and polarization of basal cell nuclei, and cytoplasmic vacuolization (Fig. 8, A). Although the cyst-lining epithelium contained a considerable number of ghost cells and calcification (Fig. 8, B), the transformed ameloblastomatous epithelial portion showed little or no ghost cells and calcification. Juxtaepithelial dentinoid was not present. This lesion could be differentiated from ameloblastomatous COC by the histopathologic criteria of early ameloblastoma suggested by Vickers and Gorlin, the lack of ghost cells and calcification in the transformed ameloblastoma areas, and extralu- minal growth.

I I I

I 37/M Anterior mandibular Small. soft mass gingiva

2 61/M Mandibular edentulous Pedunculated mucosa soft mass*

3 74/F Mandibular edentulous Slowly growing, mucosa small red nodule

4 76/M Mandibular edentulous Asymptomatic nodular mucosa swelling

5 49/F Nonspecified edentulous Yellowish white mucosa mucosal plaque*

6 50/F Mandibular edentulous Red papillomatous mucosa lesion*

7 79/M Mandibular edentulous Painless friable mucosa enlargement for

3-6 mo* 8 69/F Mandibular edentulous Slightly raised,

mucosa painful swelling*

*Denture wearer.

Because only two cases were found, the clinical features of this group could not be clearly defined. They occurred in a lo-year-old girl and in a 59-year-old man. Both tumors occurred in the posterior of the mandible and appeared as intraosseous, well-defined radi- olucent lesions that produced progressive swelling.

Eight of the 13 neoplastic lesions were located in the extraosseous gingival or alveolar mucosa and bore a striking resemblance to peripheral ameloblastoma except that ghost cells were clustered in the central portion of the tumor cell nests and juxtaepithelial dentinoid was adjacent to the most peripheral cells (Fig. 9). These lesions were not encapsulated. Multi- focal downward proliferation of the mucosal epithelium was usually noted. Clusters of neoplastic odontogenic epithelium with central ghost cells were in a fibromyxoid stroma. The basal epithelial cells were frequently atrophic, and, in contrast to ameloblastoma, palisading of basal cell nuclei was lacking. For- eign body granulomas and calcification of ghost cells were not prominent. Conforming to the terminology of Ellis and Smookler, ** these lesions were designated as peripheral epithelial odontogenic ghost cell tumors.

The patients’ ages ranged from 37 to 79 years, with a mean age of 62 years; six patients were more than 50 years old. There was no sex predilection. In seven patients these tumors occurred in edentulous regions, and five of these patients were known to have used re- movable dental prostheses. The clinical appearance of these soft tissue tumors was variously described as exophytic and pedunculated, nodular, slightly raised, and plaquelike with a hard, soft, or friable consistency (Table II).

Volume 12 Number I

Calcifying odontogenic cyst 6 1

The other three tumors were intraosseous and characterized by nests or clusters of proliferative odontogenic epithelium that contained ghost cells and calcifications (Fig. 10). Juxtaepithelial dentinoid or dysplastic dentin was usually prominent but variable in quantity from lesion to lesion (Fig. 10, A). Under low magnification these tumors resembled ameloblastoma or adenomatoid odontogenic tumor. In some areas neoplasic cells tended to form cribriform and tubular patterns. Ghost cells were usually encoun- tered in the midst of epithelial clusters and rarely found in the connective tissue (Fig. 10, B). Foreign body granulomas due to herniated ghost cells were rare. These lesions were designated central epithelial odontogenic ghost cell tumors.

These tumors occurred in male patients of ages 12, 49, and 75 years. Radiographs of these tumors showed multilocular radiolucencies in the mandible and a radiopaque mass that extended into the maxillary sinus. The mandibular lesion in the 49-year-old man was associated with an impacted premolar tooth. These tumors were slowly growing, expansile masses, but both mandibular tumors had exhibited sudden in- creases in size.

lmmunohistochemistry

The peroxidase-antiperoxidase staining method with polyclonal antikeratin antibody revealed that ghost cells were unreactive or weakly immunostained in contrast to the intense immunostaining of adjacent odontogenic epithelium.

DISCUSSION

The term calcifying odontogenic cyst was first suggested by Gorlin et al. 4, 5 Although other terms have been presented to designate this lesion,‘-3, 6-9 COC is a suitable term to designate these cystic lesions of odontogenic origin with calcifying potential.

COC has been histopathologically defined by the World Health Organization as a nonneoplastic cystic lesion that is lined by enamel organlike epithelium, contains denucleated eosinophilic ghost cells and calcification in the epithelium and connective tissue wall, and is sometimes associated with other features.21 Although ghost cell formation can be seen in several odontogenic and nonodontogenic lesions,’ ‘3 22-27 it is characteristic of COC.28, 29

Many investigators have made efforts to clarify the nature of ghost cells by employing special histochem- ical methods, transmission electron microscopy, and scanning electron microscopy, and various theories have been proposed without any general agreement. Gorlin et al.,4, 5 Ebling and Wagner,30 Gold,6 Bhaskar,7 Komiya et al.,31 and Regezi et al.26 all believed that ghost cells represent normal or abnormal keratinization. Levy23 investigated ghost cells in od-

Fig. 10. Central epithelial odontogenic ghost cell tumor exhibits proliferation of epithelium without cyst lumen in pattern similar to ameloblastoma or adenomatoid odonto- genie tumor, but juxtaepithelial dentinoid (A) and ghost cell formations (B) are present. (Hematoxylin-eosin stain; original magnification, X 150.)

ontomas and suggested that they represent squamous metaplasia with subsequent calcification caused by ischemia. This theory was supported by Sedan0 and Pindborg22 and by Kerebel and Kerebel.25 Sedan0 and Pindborg thought the ghost cells represented different stages of normal and aberrant keratin formation and that they were derived from the metaplastic transformation of odontogenic epithelium. Other investigators suggested or implied that ghost cells may represent the product of abortive enamel matrix in odontogenic epithelium. 32 However, the morphology of ghost cells seems different from that of enamel matrix.

Peroxidase-antiperoxidase immunohistochemical analysis with polyclonal cytokeratin antibody performed in this study demonstrated that most typical denucleated ghost cells reacted very faintly or not at all with polyclonal cytokeratin antibody, in contrast to the marked reaction of adjacent odontogenic epithe-

62 Hong, Ellis, and Hartman ORAL SURG ORAL MED ORAL PATHOL July 1991

lium. The weak immunoreaction of ghost cells sug- gests that the keratin antigens have been altered.

Ghost cells are characterized by the loss of nuclei, clear preservation of basic cellular outlines, resistance to resorption, induction of foreign body granulomas, and the potential to calcify. In addition, in formalin- fixed tissue they express little or no cytokeratin immunoreactivity. These characteristics are compat- ible with the features of coagulative necrosis.33 There- fore ghost cells may represent the product of coagulative necrosis of odontogenic epithelium.

It is established that several types of odontogenic cyst develop as a result of central liquefaction necrosis of proliferative epithelial clusters or through proliferation of epithelium to line the connective tissue cavity formed by focal necrosis of connective tissue.34 We suggest that in the case of COC, central liquefaction necrosis.of odontogenic epithelial clusters may take place in the initiation phase of cyst development, and coagulative necrosis occurs at the same time or later in portions of the cyst-lining epithelium, with resultant ghost cell formation. As the evolution or development of COC proceeds, isolated or clustered ghost cells calcify.

Calcification in various forms in the epithelium and connective tissue wall is another peculiar feature of COC. Abrams and Howel13s suggested that the ghost cells induce adjacent granulation tissue to lay down juxtaepithelial osteoid that may calcify. Sauk,36 Na- gao et a1.,14 and Sapp and Gardner37 contended that the juxtaepithelial osteoid or dentinoid of COC is due to a true inductive phenomenon rather than an inflammatory response. We found juxtaepithelial dentinoid that was not associated with granulation tissue as well as dystrophic calcification in clustered ghost cells.

There has been universal agreement on the odonto- genie origin of COC since Gorlin et a1.4, s suggested it. They pointed out the histopathologic similarities of basal layer cells to ameloblastic cells, the production of dentinoid, and the exclusive occurrence in locations with a close relationship to dental structures.

We divided 92 cases in this study into two basic entities: 79 (85%) cysts and 13 (14%) neoplasms. The classification system of so-called COC (Table I) is based on the proliferative activity and growth pattern of the cyst-lining epithelium, the microscopic pattern of cyst formation, and clinical features.

Nonproliferative and proliferative cysts occurred both intraosseously and extraosseously. Vuletin et a1.38 reported a unusual case under the name “peripheral odontogenic tumor with ghost cell keratinization.” It occurred in a 3-year-old black girl. Their photomicrograph illustrated features of the nonproliferative variant of COC.

We speculate that when cystic degeneration is

greater than the proliferative activity of the cyst- lining epithelium, nonproliferative COC (type IA) will develop. When the proliferative activity of the epithelium exceeds cystic degeneration, proliferative COC (type IB) is formed. In contrast to the proliferative COC, nonproliferative COC has a simple unicystic structure, lack of proliferative activity of the cyst-lining epithelium, and lack of multiple daughter cysts in the connective tissue wall. Proliferative COC has more extensive ghost cell formation and a greater tendency to calcify. An immature odontoma can be distinguished from proliferative COC by an absence of prominent ghost cell formation in the central portion of the odontogenic epithelium, evidence of tooth formation and juxtaepithelial dentinoid, and close contact with dental papilla-like myxoid tissue.

Ameloblastomatous COC (type IC) microscopi- cally resembles unicystic ameloblastoma except for the ghost cells and calcification within the proliferative epithelium. Ameloblastomatous COC occur only intraosseously. This subtype of COC is distinct from true ameloblastoma arising in COC. In contrast to ameloblastoma ex COC, the ghost cells and dystrophic calcification are within the proliferative epithelium, which lacks histopathologic criteria suggested by Vickers and Gorlin20 and is confined to the cyst lumen. It is differentiated from the benign epithelial odontogenic ghost cell tumor by its obvious cystic structure and lack of juxtaepithelial dentinoid.

Type ID COC showed the combined features of nonproliferative COC and odontoma. Praetorius et al.” believed the odontoma developed in the COC. We suggest that these COCs develop in a manner similar to that of dentigerous cysts; that is, the COC develops as a result of the odontoma.

Ameloblastoma ex COC (type IIA) designates ameloblastoma arising from the cyst-lining epithelium of COC. There have been numerous reports of ameloblastoma arising from odontogenic cysts, in- cluding dentigerous cyst, primordial cyst, radicular cyst, and residual cyst.39-46 However, review of the English-language literature failed to reveal a case of ameloblastoma arising from COC. We found two ex- amples in our series. Ameloblastoma ex COC occurred only intraosseously, appearing as cystlike, ra- diolucent lesions in the posterior mandible. Whether these tumors have the same destructive potential and propensity for recurrence as typical ameloblastomas is unknown.

We have used the term “epithelial odontogenic ghost cell tumor” to designate the other neoplastic variant of COC. Praetorius et al.’ ’ suggested the term “dentinogenic ghost cell tumor.” The term dentino- genie seems to connote a mesenchymal tissue origin and the production of true dentin. We believe that the characteristic features of these neoplasms are the od-

Volume 72 Number 1


ontogenic epithelial proliferation with some inductive activity and the formation of ghost cells.

Epithelial odontogenic ghost cell tumor was divided into two different entities, peripheral and central, on the basis of clinicopathologic features. In addition, Ellis and Smookleri2 described aggressive or malignant epithelial odontogenic ghost cell tumors that showed aggressive growth and malignancy-associated histologic features.

The peripheral epithelial odontogenic ghost cell tumor (type IIB) has been first described in this study. This tumor resembles the peripheral ameloblastoma except for ghost cell formations in the central portion of neoplastic epithelial clusters and juxtaepithelial dentinoid. They occurred predominantly as nodular swellings on the edentulous alveolar mucosa of denture wearers, a feature that implicates trauma or ir- ritation as etiologic factors.

A similar case of peripheral epithelial odontogenic ghost cell tumor appeared in Fejerskov and Krogh’s report.8 They described a broad-based mass on the palatal mucosa of a denture wearer. The published photomicrograph, histologic description, and clinical features are in common with the features outlined here for this tumor.

Three cases of intraosseous epithelial odontogenic ghost cell tumor (type IIC) were found in this study. The histopathologic features of this tumor varied from area to area. Some portions resembled ameloblastoma-like epithelium, whereas other areas showed adenomatoid odontogenic tumor-like features with a cribriform or tubular pattern. The common histologic findings were ghost cells and dentinoid; however, the tumor cells lacked the ameloblastic histopathologic criteria suggested by Vickers and Gorlin.20 Some tumors showed central cystic degeneration. These tumors appeared as expansile, multilocular radiolucen- ties with focal radiopacities. One of them occurred in a patient with the Gardner syndrome.

REFERENCES

1. Rywkind AW. Beitrag zur Pathologie der Cholesteatome. Virchows Arch [A] 1932;28:3 13-28.

2. Thoma KH, Goldman HM. Odontogenic tumors: a classification based on observations of the epithelial, mesenchymal and mixed varieties. Am J Path01 1946;12:433-7 1.

3. Maitland GR. Atypical adamantinoma of the maxilla: report of case. J Oral Surg 1947;5:351-5.

4. Gorlin RJ, Pindborg JJ, Clausen FP, Vickers RA. The calcifying odontogenic cyst: a possible analogue of the cutaneous calcifying epithelioma of Malherbe-an analysis of fifteen cases. ORAL SURG ORAL MED ORAL PATHOL 1962;15:1235- 43.

5. Gorlin RJ, Pindborg JJ, Redman RS, Williamson JJ, Hansen LS. The calcifying odontogenic cyst: a new entity and possible analogue of the cutaneous calcifying epithelioma of Malherbe. Cancer 1964;17:723-9.

6. Gold L. The keratinizing and calcifying odontogenic cyst. ORAL SURC ORAL MED ORAL PATHOL 1963;16:1414-24.

7. Bhaskar SN. Oral Surgery-Oral Pathology Conference No. 13, Walter Reed Army Medical Center: gingival cyst and the keratinizing ameloblastoma. ORAL SURG ORAL MED ORAL PATHOL 1965;19:796-807.

8. Fejerskov D, Krogh J. The calcifying odontogenic tumor or the calcifying odontogenic cyst. J Oral Path01 1972;1:273-87.

9. Freedman PD, Lumerman H, Gee JK. Calcifying odontogenic cyst: a review and analysis of seventy cases. ORAL SURG ORAL MED ORAL PATHOL 1975;40:93-105.

10. Praetorius F. Calcifying odontogenic cyst: range, variations and neoplastic potential. Int J Oral Surg 1975;4:89.

11. Praetorius F. Hiortinn-Hansen E. Gorlin RJ. Vickers RA. Calcifying odoniogenic cyst: range, variations and neoplastic potential. Acta Odontol Stand 1981;39:227-40.

12. Ellis GL, Smookler BM. Aggressive (malignant?) epithelial odontogenic ghost cell tumor. ORAL SURG ORAL MED ORAL PATHOL 1986;6 1:47 l-8.

13. Nagao T, Nakajima T, Fukushima M, Ishiki T. Calcifying odontogenic cyst. J Maxillofac Surg 1983;11:174-9.

14. Nagao T, Nakajima T, Fukushima M. Ishiki T. Calcifying odontogenic cyst with complex odontoma. J Oral Maxillofac Surg 1982;40:810-3.

15. Farman AG, Smith SN, Norje CJ, Grotepass FW. Calcifying odontogenic cyst with ameloblastic fibrocdontoma: one lesion or two? J Oral Pathol 1978;7:19-27.

16. Zachariades N. Unusual Gorlin cyst with traumatic neuroma. J Oral Med 1985;40:87-90.

17. Wright BA, Bhardwaj AK, Murphy D. Recurrent calcifying odontogenic cyst. ORAL SURG ORAL MED ORAL PATHOL 1984;58:579-83.

18. Ikemura K, Horie A, Tashiro H, Nandate M. Simultaneous occurrence of calcifying odontogenic cyst and its malignant transformation. Cancer 1985;56:2861-4.

19. Shafer WG, Hine MK, Levy BM. A textbook of oral pathology. 4th ed. Philadelphia: WB Saunders, 1983:274-5.

20. Vickers RA, Gorlin RJ. Ameloblastoma: delineation of early histopathologic features of neoplasia. Cancer 1970;26:699- 710.

21. Pindborg JJ, Kramer IRH, Torloni H. Histologic typing of odontogenic tumors, jaw cysts and allied lesions. Geneva: World Health Organization, 1972:28.

22. Sedan0 HO, Pindborg JJ. Ghost cell epithelium in odontomas. J Oral Pathol 1975;4:27-30.

23. Levy BA. Ghost cells and odontomas. ORAL SURC ORAL MED ORAL PATHOL 1973;36:851-5.

24. Regezi JA, Courtney RM, Kerr DA. Keratinization in odontogenic tumors. ORAL SURC ORAL MED ORAL PATHOL 1975;39:447-55.

25. Kerebel B, Kerebel L. Ghost cells in complex odontoma: a light microscopic and SEM study. ORAL SURG ORAL MED ORAL PATHOL 1985;59:371-8.

26. Regezi JA, Kerr DA, Courtney RM. Odontogenic tumors: analysis of 706 cases. J Oral Surg 1978;36:771-8.

27. Bernstein ML, Buchino JJ. The histologic similarity between craniopharyngioma and odontogenic lesions: a reappraisal. ORAL &RG ORAL MED ORAL FATHOL 1983;56:502:il.

28. Ng KH, Siar CH. Clear cell change in a calcifying odonto- genie cyst. ORAL SURG ORAL MED ORAL PATHOL 1985;60: 417-9. -

29. Soams JV. A pigmented calcifying odontogenic cyst. ORAL SURG ORAL MED ORAL PATHOL 1982;53:395-400.

30. Ebling H, Wagner JE. Calcifying odontogenic cyst. ORAL SURG ORAL MED ORAL PATHOL 1967;24:537-9.

3 1. Komiya Y, Susa A, Kawachi H, Yamamura T, Eda S. Calci- fying odontogenic cyst: report of a case. ORAL SURG ORAL MED ORAL PATHOL 1969;27:90-4.

32. David R, Buchner A. Calcifying odontogenic cyst with intra- cellular amyloid-like material. ORAL SURG ORAL MED ORAL PATHOL 19?‘6;41:758-64.

33. Robbins SL. Cotran RS. Patholoaic basis of disease. 2nd ed. Philadelphia: WB Saunders, 197’$36-7.

34. Shafer WG, Hine MK, Levy BM. A textbook of oral pathology. 4th ed. Philadelphia: WB Saunders, 1983:494.

35. Abrams AM, Howell FV. The calcifying odontogenic cyst: re-

64 Hong, Ellis, and Hartman

36.

31.

38.

39.

40.

41.

42.

port of four cases. ORAL SURC ORAL MED ORAL PATHOL 1968;25:594-606. Sauk JJ. Calcifying and keratinizing odontogenic cyst. J Oral Surg 1972;30:893-7. Sapp JP, Gardner DC. An ultrastructural study of the calcifications in calcifying odontogenic cysts and odontomas. ORAL SURG ORAL ME; URAL PA~HOL 1!977;44:754-66. Vuletin JC. Solomon MP. Pertschuk LP. Peripheral odonto- genie tumo; with ghost-ceil keratinization. ORAL SURG ORAL MED ORAL PATHOL 1978;45:406-15. Small GS, Lattner CW, Waldron CA. Ameloblastoma of the mandible simulating a radicular cyst. J Oral Surg 1958;6: 231-5. Stanley HR, Diehl DL. Ameloblastoma potential of follicular cysts. ORAL SURG ORAL MED ORAL PATHOL 1965;20:260-8. Lee FMS. Ameloblastoma of the maxilla with possible origin in a residual cyst. ORAL SURG ORAL MED ORAL PATHOL 1970;29:799-805. Byrd DL, Allen JW, Dunsworth AR. Ameloblastoma origi- nating in the wall of a primordial cyst. ORAL SURG ORAL MED ORAL PATHOL 1973;3 1:301-4.

43.

44.

45.

46.

ORAL SURG ORAL MED ORAL PATHOL July 1991

Generson RM, Porter JM, Stratigos GT. Mural odontogenic epithelial proliferations within the wail of a dentigerous cyst: their significance. ORAL SURC ORAL MED ORAL PATHOL 1976;42:717-21. Shteyer A, Lustmann J, Lewin-Epstein L. The mural ameloblastoma: a review of the literature. J Oral Surg 1978;36:866- 72. McMillan MD, Smillie AC. Ameloblastomas associated with dentigerous cysts. ORAL SURG ORAL MED ORAL PATHOL 1981;51:489-96. Leider AS, Eversole LR, Barkin ME. Cystic ameloblastoma: a clinicopathologic analysis. ORAL SURG ORAL MED ORAL PATHOL 1985;60:624-30.

Reprint requests: Armed Forces Institute of Pathology Editorial Office (AFIP-ZSP-E) Washington, DC 20306-6000

gorlin´s cisty

Documents