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Clear cell change in a calcifying odontogenic cyst

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Clear cell change in a calcifying odontogenic cyst K. H. Ng, B.D.S., M.Sc., F.D.S.R.C.P.S.,* and C. H. Siar, B.D.S., M.Sc., F.D.S.R.C.P.S.,** Kuala Lumpur, Malaysia A case of calcifying odontogenic cyst exhibiting clear cell change is described. Histomorphologic similarities to the clear cell type of calcifying epithelial odontogenic tumor were noted and the importance of differential diagnosis from clear cell types of salivary gland neoplasms and metastatic hypernephroma are emphasized. (ORAL. SURG. ORAL MED. ORAL PATHOL. 60~417-419, 1985) T he calcifying odontogenic cyst (COC) was first characterized by Gorlin and his associates’ in 1962 and is now recognized as a distinct entity by the World Health Organization.* Since its inception more than 150 caseshave been reported and several reviews have been documented. The COC is generally regarded as a benign lesion of odontogenic origin, which can present as a cyst or as a neoplasm.3 Praetorius and co-authors) classified the COC into two main subentities: a unicystic type and a neoplastic type. They proposed that the latter should be called dentinogenic ghost cell tumor. It is well-recognized that the COC often occurs in association with other lesions such as odontomas.4 A pigmented variant has also been reported.5 Clear cell change in a COC has not been previously docu- mented in the literature. The purpose of this article is to describe a case that exhibited this unusual change. CASE REPORT A 29-year-old Malay woman was first seen with a chief complaint of a swelling involving the right side of the mandible. This extended from the mandibular first premo- lar to the mandibular second molar. The preliminary clinical diagnosis was radicular cyst. Enucleation of the cyst and curettage were performed, and the cyst lining was sent for microscopic examination. *Head, Division of Stomatology, Institute for Medical Research. Jalan Pahang. **Lecturer, Department of Oral Pathology and Oral Medicine, Faculty of Dentistry, University of Malaya. Macroscopic findings The surgical specimen consisted of a piece of cyst lining measuring approximately 3.5 X 2.4 X 0.8 cm. Microscopic findings The specimen was fixed in formalin and processed in the routine manner. Paraffin sections were prepared and initially stained with hematoxylin and eosin. Subsequent sections were stained with alcian blue, Southgate mucicar- mine, and PAS-diastase. Microscopically, the specimen consisted of a fibrous connective tissue wall lined with stratified squamous epi- thelium. The epithelium had a well-defined basal cell layer composedof columnar cells that exhibited polarization of their nuclei away from the basement membrane. Superfi- cial to this was a loosely structured network of stellate reticulum-like cells and focal masses of ghost cells undergoing varying degrees of calcification (Fig. 1). The histologic appearance conformed to that necessary for a diagnosis of calcifying odontogenic cyst as defined by the World Health Organization Committee for the Histo- logical Typing of Odontogenic Tumors, Jaw Cysts and Allied Lesions.* An unusual finding was the presence of nests, cords, and islands of large polyhedral cells in the subepithelial zone of the connective tissue wall (Fig. 2). These cells had clear or finely granular cytoplasm with small ovoid nuclei often displaced to the periphery. Stains for mucin and glycogen were negative. These clumps of cells were sometimes closely related to areas of hyaline material or dentinoid which were present in a subepithelial location (Fig. 3). DISCUSSION A review of the English-language literature revealed that clear cell change in the COC has not 417
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Clear cell change in a calcifying odontogenic cyst K. H. Ng, B.D.S., M.Sc., F.D.S.R.C.P.S.,* and C. H. Siar, B.D.S., M.Sc., F.D.S.R.C.P.S.,** Kuala Lumpur, Malaysia

A case of calcifying odontogenic cyst exhibiting clear cell change is described. Histomorphologic

similarities to the clear cell type of calcifying epithelial odontogenic tumor were noted and the importance

of differential diagnosis from clear cell types of salivary gland neoplasms and metastatic hypernephroma

are emphasized. (ORAL. SURG. ORAL MED. ORAL PATHOL. 60~417-419, 1985)

T he calcifying odontogenic cyst (COC) was first characterized by Gorlin and his associates’ in 1962 and is now recognized as a distinct entity by the World Health Organization.* Since its inception more than 150 cases have been reported and several reviews have been documented.

The COC is generally regarded as a benign lesion of odontogenic origin, which can present as a cyst or as a neoplasm.3 Praetorius and co-authors) classified the COC into two main subentities: a unicystic type and a neoplastic type. They proposed that the latter should be called dentinogenic ghost cell tumor.

It is well-recognized that the COC often occurs in association with other lesions such as odontomas.4 A pigmented variant has also been reported.5 Clear cell change in a COC has not been previously docu- mented in the literature. The purpose of this article is to describe a case that exhibited this unusual change.

CASE REPORT

A 29-year-old Malay woman was first seen with a chief complaint of a swelling involving the right side of the mandible. This extended from the mandibular first premo- lar to the mandibular second molar. The preliminary clinical diagnosis was radicular cyst. Enucleation of the cyst and curettage were performed, and the cyst lining was sent for microscopic examination.

*Head, Division of Stomatology, Institute for Medical Research. Jalan Pahang. **Lecturer, Department of Oral Pathology and Oral Medicine, Faculty of Dentistry, University of Malaya.

Macroscopic findings

The surgical specimen consisted of a piece of cyst lining measuring approximately 3.5 X 2.4 X 0.8 cm.

Microscopic findings

The specimen was fixed in formalin and processed in the routine manner. Paraffin sections were prepared and initially stained with hematoxylin and eosin. Subsequent sections were stained with alcian blue, Southgate mucicar- mine, and PAS-diastase.

Microscopically, the specimen consisted of a fibrous connective tissue wall lined with stratified squamous epi- thelium. The epithelium had a well-defined basal cell layer composed of columnar cells that exhibited polarization of their nuclei away from the basement membrane. Superfi- cial to this was a loosely structured network of stellate reticulum-like cells and focal masses of ghost cells undergoing varying degrees of calcification (Fig. 1).

The histologic appearance conformed to that necessary for a diagnosis of calcifying odontogenic cyst as defined by the World Health Organization Committee for the Histo- logical Typing of Odontogenic Tumors, Jaw Cysts and Allied Lesions.*

An unusual finding was the presence of nests, cords, and islands of large polyhedral cells in the subepithelial zone of the connective tissue wall (Fig. 2). These cells had clear or finely granular cytoplasm with small ovoid nuclei often displaced to the periphery. Stains for mucin and glycogen were negative. These clumps of cells were sometimes closely related to areas of hyaline material or dentinoid which were present in a subepithelial location (Fig. 3).

DISCUSSION

A review of the English-language literature revealed that clear cell change in the COC has not

417

4 I a Ng and Siar Oral Surg. October, 1985

Fig. 1. Areas showing classic features of a calcifying odontogenic cyst. (Hematoxylin and eosin stain. Original magnification, X200.)

Fig. 2. Nests, cords, and islets of clear cells in the subepithelial connective tissue zone. (Hematoxylin and eosin stain. Original magnification, X40.)

been previously described. However, four cases of calcifying epithelial odontogenic tumor (CEOT) exhibiting clear cell change have been reported.6-8 Certain histomorphologic similarities were noted between the clear cells in CEOT and COC. In both lesions, the clear cells were large and irregularly ovoid with cytoplasm that varied from a finely granular, through a loosely reticular, to a clear type, depending on the degree of vacuolization.8 These cells characteristically formed clusters in an orga-

noid configuration with a closely supportive and well-organized collagenous stroma.

The clear cell variation in COC and CEOT could pose problems in diagnosis. The glandular configura- tion of these cells could prompt an incorrect diagno- sis of clear cell variants of salivary gland neoplasm5 and metastatic hypernephroma.’ This is particularl} true when small incisional biopsy specimens an submitted for histologic confirmation.

The nature of these clear cells in COC and CEO:

Volume 60 Number 4

Clear cell change in calcifying odontogenk cyst 419

Fig. 3. Hyalinized stromal collagen and a zone of dentinoid between epithelial lining and clear cells.

(Hematoxylin and eosin stain. Original magnification, X 100.)

is still not known. They are probably odontogenic epithelial cells that have undergone aberrant degen- eration.

REFERENCES

I. Gorlin RJ, Pindborg JJ, Clausen FP, Vickers RA: The calcifying odontogenic cyst-a possible analogue of the cuta- neous calcifying epithelioma of Malherbe: an analysis of fifteen cases. ORAL SURC ORAL MED ORAL PATHOL 15:1235- 1243. 1962.

2. Pindborg JJ, Kramer IRH, Torloni H: Histological typing of odontogenic tumours, jaw cysts and allied lesions. No. 5. International Histologic Classification of Tumours Series, Geneva, 1971, World Health Organization, p. 28.

3. Praetorius F. Hjorting-Hansen E, Gorlin RJ, Vickers RA: Calcifying odontogenic cyst: range, variation and neoplastic potential. Acta Odontol Stand 39: 227-240, 1981,

4. Nagao T, Nakajima T, Fukushima M, lshiki T: Calcifying odontogenic cyst: a survey of 23 cases in the Japanese literature, J Maxillofac Surg 11: 174-179, 1983.

Soames JV: A pigmented calcifying odontopcnic cyst. OK,\I SURC ORAL MED ORAL PATHOL 52: 395-400, 1982. Abrams AM, Howell FV: Calcifying epithelial odontogenic tumour: report of four cases. J Am Dent Assoc 74: I23 I - 1240, 1967. Franklin CD, Pindborg JJ: The calcifying epithelial odonto- genie tumour: a review and analysis of I 13 (cases. ORAI SL~KG ORAL MED ORAL PATHOL 42: 753-765, 1976. Greer RJ. Richardson JF: Clear-cell calcifying odontogenic tumour viewed relative to the Pindborg tumour. 0~41 SLKG ORAL MED ORAL PATHOI. 42: 775-779. 1976

Reprint requests to:

Dr. K.H. Ng Division of Stomatology Institute for Medical Research Jalan Pahang Kuala Lumpur, Malaysia


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