resorption of the bone crest between the right mandibular lateral incisor and canine

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口腔病理科 On-Line KMU Student Bulletin

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原文題目(出處)： Clear Cell Variant of Calcifying Epithelial Odontogenic Tumor: Is It Locally Aggressive?

J Oral Maxillofac Surg 2009;67:207-11.

原文作者姓名： Ana Lúcia Carrinho Ayrosa Rangel, Andréia Aparecida da Silva, Fábio Augusto Ito, Márcio Ajudarte Lopes, Oslei Paes de Almeida, DDS, PhD, Pablo Agustin Vargas

通訊作者學校： Piracicaba Dental School, State University of Campinas, Piracicaba, São Paulo, Brazil.

報告者姓名(組別)： Intern L組陳品卉 報告日期： 98.02.13

內文：

The calcifying epithelial odontogenic tumor (CEOT) is a rare benign odontogenic neoplasm of the jaws, accounting for approximately 1% of all intraosseous odontogenic tumors.

Some histologic variants have been described, including CEOT with Langerhans cells, with cementum-like and bone-like material, combined epithelial odontogenic tumor and adenomatoid odontogenic tumor, myoepithelial cells, and the clear cell variant of calcifying epithelial odontogenic tumor (CCCEOT).

The diagnosis of CCCEOT is very difficult and other clear cell lesions that affect the oral cavity should be excluded. According to some authors it is more aggressive when compared with other CEOT variants showing a recurrence rate of 10.52% and should be considered as a low-grade odontogenic carcinoma.

Report of a Case

a specimen from a 65-year-old male of a painless swelling located between the mandibular right lateral incisor and canine that had been growing slowly for about 3 years.

Clinical information Æ a fibrous, firm, sessile, and painless lesion covered by a smooth and regular mucosal surface in the anterior mandibular gingival between the right lateral incisor and canine, which measured nearly 1.5 cm in its maximum dimension.

Periapical radiograph Æ a well-defined unilocular radiolucent lesion with radiopacities dispersed throughout the lesion.

resorption of the bone crest between the right mandibular lateral incisor and canine; however, the roots of the adjacent teeth were not displaced

no sign of the root resorption (Fig 1).

The lesion was removed by complete surgical excision.

Histolopathologic Finding

Dense connective tissue that was replaced by irregular

strands, cords, and nests of epithelial cells.

The poly hedr al epit heli al cells disclosed an abundant and

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eosinophilic cytoplasm; round to oval relatively large nuclei with dense chromatin, and evident intercellular bridges. These cells showed mild pleomorphic and hyperchromatic nuclei.

These cells showed mild pleomorphic and hyperchromatic nuclei. In significant portions, the epithelial cells had a clear, foamy, vacuolated cytoplasm(Figs 2-4).

A: The calcifications areas and Liesegang’s rings were observed

B: Amyloid-like deposits could be observed in Congo red staining under polarized light (Fig5). Fig. 6: avidin-biotin complex technique showed positivity for cytokeratin cocktail in the epithelial neoplastic cells

Fig.7: the stromal component was positive for vimentin

Analyzing all features described above the final diagnosis of central CCCEOT was established. The patient has been followed for about 24 months with no sign of recurrence observed.

Discussion

Currently 20 reported cases, 12 central and 8 peripheral lesions.

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- 3 - M:F ratio Æ central 1:1.2 ; peripheral 1.66:1.

Age ranged from 14 to 68 years with a mean age of 40.84 years. The mean age for the intraosseous CCCEOT is higher (46.36 years) than for the extraosseous variant (33.25 years).

The surgical management modalities in the 20 cases were as follows: complete or partial resection, excision, enucleation, and curettage.

The follow-up ranged from 4 months to 13 years (mean 3.6 years), and it was not available in 1 case. Recurrence was reported in 2 cases of central CCCEOT (10.52%).

None of the peripheral lesions recurred.

Histopathological diagnosis

typical epithelial clear cells within the tumor

nuclei vary in size and shape, with rare mitotic figures

irregular strands, cords, and nests of the polyhedral epithelial cells with abundant, eosinophilic cytoplasm, round to oval relatively large nuclei with dense chromatin, and evident intercellular bridging in association with clear epithelial cells

Deposition of extracellular amyloid-like material and calcifications are also typical.

Differential Diagnosis of Clear cell tumors Epithelial lining/ clear cell

rests of dental lamina

Lateral periodontal cyst, gingival cyst

Odontogenic epithelial tumor

Ameloblastoma, calcifying odontogenic cyst, calcifying odontogenic tumor

minor degree of atypia, good circumscription of the lesion, and presence of calcified and amyloid-like material.

Salivary gland tumors mucoepidermoid carcinoma, acinic cell carcinoma

Actin, S-100 (-), PAS (+) metastatic disease kidney, thyroid, and lung

carcinomas

PAS (-) melanocytic and

mesenchymal neoplasms.

arise in the soft tissue, rarely in the head and neck region.

S-100 (+)

melanoma associated antigen (HMB-45) (+)

The diagnosis of CCCEOT in our case was supported by microscopically biphasic pattern, presence of apple-green birefringent/ Congo red-positive material between tumor islands, small calcifications, good circumscription of the lesion, and lack of mitotic figures.

Our analysis showed that all cells (polyhedral and clear) strongly expressed CK AE1/AE3, CK7, and CK14 but were immunonegatives for S-100, muscle specific actin, desmin, and anti-human melanosome. The stromal component, however, was positive for vimentin.

Although considered benign in nature, CCCEOT is designed as locally aggressive for some investigators because its moderate recurrence rate.

Anavi et al: there is evidence to support the classification of CCCEOT as a distinct, more aggressive variant of CEOT but not as a separate entity.

Veness et al described a case of CEOT suggesting a malignant transformation process.

Kumar et al reported a case with initial diagnosis of CEOT that showed posterior widespread infiltration, necrosis, and metastatic lesion in vertebrae and hip. Final diagnosis was metastasizing clear cell odontogenic carcinoma.

Treatment of CCCEOT involves surgery; enucleation of involved tooth, or “in bloc resection” in some cases including any associated soft tissue mass. Tumor-free surgical margins should be obtained to reduce the risk of local recurrences. The recurrent cases of

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CCCEOT occurred probably because of inadequate treatment.

The clinical course of the present case did not show recurrence after 24 months.

CCCEOT is not locally aggressive as mentioned previously because the cases reported have generally shown little evidence of aggressiveness even years after relatively conservative treatment.

題號題目

1 下列有關adenomatoid odontogenic tumor, calcifying odontogenic tumor及 ameloblastic fibro-odontoma三者共同點的敘訴，何者錯誤?

(A) 都含有來自外胚層(ectoderm)的組織

(B) 在放射線影像上都表現出同時有放射線透射及不透射區

(C) 三者都是齒源性的良性腫瘤

(D) 都比較常見於50歲以上的病患

答案( D ) 出處：94年第二次高考

AOT 好發在10~19歲的年輕人身上; calcifying odontogenic tumor 平均年 齡17歲； ameloblastic fibro-odontoma 常見於小孩，平均年齡約10歲左 右。

Neville’s Oral and Maxillofacial Pathology, 2^nd Ed.

題號題目

2 在組織病理切片裡，鈣化性齒源性囊腫(calcifying odontogenic cyst)最特別

的是有下列何者?

(A) cyst (B) ghost cells (C) stellate reticulum (D) Inflammation 答案( B ) 出處：96年第一次高考

“The most characteristic histoathologic feature of the calcifying odontogenic cyst is the presence of variable numbers of ghost cells within epithelial component.”

Neville’s Oral and Maxillofacial Pathology, 2^nd Ed. (pg. 605) 題號題目

3 關於calcifying epithelial odontogenic tumor，下列何者為非?

(A) 常見於30~50歲的中年人

(B) 病理玻片下可看到Lisesgang’s rings以及鈣化物質

(C) 在放射線影像上整個病灶區是完全不透射區

(D) 在congo red的染色下可染出amyloid substance。

答案( C ) “Radiographically, the tumor shows a unilocular or, more often, a multilocular radiolucent defect.”

Neville’s Oral and Maxillofacial Pathology, 2^nd Ed. (pg. 623) 題號題目

4 下列何者不是calcifying epithelial odontogenic tumor在病理組織切片下所 觀察的結果？

(A) irregular strands, cords, and nests of the polyhedral epithelial cells (B) pleomorphic and hyperchromatic nuclei.

(C) Liesegang ring calcifications (D) Basophilic extracellular material

答案( D ) “The polyhedral epithelial cells disclosed an abundant and eosinophilic cytoplasm”

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