Adenomatoid odontogenic tumor associated with calcifying epithelial odontogenic tumor

A case of odontogenic tumor which contained areas diagnostic for both adenomatoid odontogenic tumor and calcifying epithelial odontogenic tumor arising in the upper left anterior region in a 17-year-old Japanese female is reported. The histopathological observation suggested that the lesion represented primarily adenomatoid odontogenic tumor in which multiple foci of calcifying epithelial odontogenic tumor had developed.     

Cystic variant of calcifying epithelial odontogenic tumor

The calcifying epithelial odontogenic tumor is a rare benign odontogenic neoplasm of the jaw. Clinically, calcifying epithelial odontogenic tumor manifests as an intraosseous lesion (central type) in the majority of cases (95%). Extraosseous or peripheral lesions account for less than 5% of cases. Calcifying epithelial odontogenic tumor can be associated with an impacted tooth and give a radiographic simulation of dentigerous cyst. Most calcifying epithelial odontogenic tumors are solid in nature, histopathologically, and might have few cyst-like spaces within them. However, a true cystic calcifying epithelial odontogenic tumor is a rare possibility. We describe a case of a true cystic variant of calcifying epithelial odontogenic tumor in a 30-year-old male, which to our knowledge, is only the second reported case.     

骨外型牙源性钙化上皮瘤

骨外型牙源性钙化上皮瘤极为罕见,通过复习1966～2011年英文MEDLINE与1994～2011年中文CHKD中收录的文献,仅有37例骨外型牙源性钙化上皮瘤的个案报告。根据上述资料分析,发现该瘤具有以下临床特点,就诊年龄3～71岁,平均35.8岁;男13例,女19例,男女比例为1∶1.46;肿块直径0.5～4 cm,平均1.76 cm;好发部位主要是牙龈,累及下颌与上颌牙龈的比例为1∶1;组织学特征表现与骨内型牙源性钙化上皮瘤基本相同;复发率为5.4%。因该瘤具有浅表骨侵蚀能力,应行完整的肿瘤切除结合骨面刮治术。     

Hybrid odontogenic tumor of calcifying odontogenic cyst and ameloblastic fibroma

Odontogenic tumors composed of 2 distinct types of lesions are unusual. We report an odontogenic tumor that was composed of calcifying odontogenic cyst and ameloblastic fibroma that occurred in the right posterior maxilla of a 22-year-old Korean woman. The tumor had a cystic component with an ameloblastic epithelial lining and conglomerates of so-called ghost cells, and there were deposits of dentinoid material adjacent to the cyst. These are features characteristic of calcifying odontogenic cyst. Enamel organ-like epithelial islands were observed within a dental papilla-like stroma of the cyst wall. Additionally, a solid portion of the tumor had characteristic features of ameloblastic fibroma, i.e., a myxoid cellular stroma with numerous elongated islands of ameloblastic epithelium. Ghost cell masses were found in the area of ameloblastic fibroma as well. The distribution of the ghost cells suggests that this is a hybrid lesion rather than a collision tumor.     

Tuberous sclerosis complex and a calcifying epithelial odontogenic tumor of the mandible

This article reports the case of a large calcifying epithelial odontogenic tumor in a patient with tuberous sclerosis complex. The calcifying epithelial odontogenic tumor, one of the rarest odontogenic tumors, is reviewed in detail, as is the unusual disorder of tuberous sclerosis complex.     

牙源性钙化上皮瘤2例

牙源性钙化上皮瘤（CEOT）是一种具有局部浸润性生长特点的良性牙源性上皮性肿瘤,可分为骨内型（中央型）、骨外型（外周型）两种,后者罕见。本文报道2例牙源性钙化上皮瘤,并结合文献探讨其临床、影像表现特点。     

Squamous odontogenic tumor

The clinical, radiographic, histologic and some immunohistochemical findings in a case of squamous odontogenic tumor (SOT) in a 56-yr-old woman are described. SOT appears to be of benign nature, most probably originating from the epithelial rests of Malassez.     

Combined epithelial odontogenic tumor: adenomatoid odontogenic tumor and calcifying epithelial odontogenic tumor

1. Two cases of a combined epithelial odontogenic tumor which had areas of AOT and CEOT were presented. 2. A review of the studies on histogenesis of the AOT revealed that the tumor probably consists of preameloblasts, stratum intermedium, and stellate reticulum. 3. A review of the studies on histogenesis of the CEOT revealed that the probable origin was in cells of stratum intermedium. 4. It is suggested that the present cases support the aforementioned theories of histogenesis and represent AOT's which contain foci of CEOT. 5. The suggested treatment is simple surgical excision.     

Calcifying epithelial odontogenic tumor of the mandible with cyst formation

We encountered a rare case of a calcifying epithelial odontogenic tumor with cyst formation. The patient was a 28-year-old man with swelling in the anterior mandibular region. Panoramic and intraoral imaging showed a clear, unilocular, radiolucent lesion with bony boundaries. Many small radiopacities were apparent in the radiolucent area. Clinically, the lesion was thought to represent an odontogenic tumor involving calcified bodies. Multidetector row computed tomography showed small radiopaque bodies arranged in a regular circular manner on the outer rim of the radiolucent region, which showed an attenuation value of approximately +20 Hounsfield units. This appearance strongly suggested a calcifying cystic odontogenic tumor, and the final diagnosis was a calcifying epithelial odontogenic tumor with cyst formation. According to the 2005 World Health Organization Classification of Tumours, cystic changes are not seen macroscopically in calcifying epithelial odontogenic tumors. We thus consider this to be a very rare and interesting case.     

Multifocal calcifying epithelial odontogenic tumor

The calcifying epithelial odontogenic tumor (CEOT), or Pindborg tumor, is a rare and benign odontogenic neoplasm that affects the jaw. The most common manifestation of CEOT is a unifocal or localized lesion of the involved jaw, which may appear clinically as a hard tissue swelling and radiographically as a mixed radiolucent-radiopaque mass. In this article, we present a unique case of CEOT affecting multiple sites in the maxilla and mandible of a 51-year-old white man. Though biopsy samples from all involved sites revealed similar histopathologic features consistent with CEOT, the fact that there was a multifocal presentation is an unusual phenomenon for CEOT and has never been reported. Multifocal odontogenic lesions are not typical but have been observed in conditions associated with known genetic mutations. For example, multiple odontogenic keratocysts are the most common feature of the inherited condition known as nevoid basal cell carcinoma syndrome. This case, however, is the first one to demonstrate that there may be a multifocal variant of CEOT that has not been previously recognized.     

Clear cell calcifying epithelial odontogenic tumor. A case report.

Clear cell variants of odontogenic tumors are rare. A case of clear cell calcifying epithelial odontogenic tumor of the mandible in a 38-year-old Caucasian male is described. This case is compared to 8 previously reported cases. The origin of clear cells in odontogenic tumors is still unknown. En bloc resection of the tumor is recommended.     

Non-calcifying variant of calcifying epithelial odontogenic tumor with Langerhans cells

Calcifying epithelial odontogenic tumor (CEOT) is a rare type of odontogenic tumor. The most characteristic feature of the classical CEOT is the presence of amyloid globules and Liesegang ring calcification in the tumor tissue. Here, we present a non-calcifying variant of intraosseous CEOT with the presence of Langerhans cells within tumor epithelial nests in a 52-year-old Taiwanese woman. The patient was referred from a local dentist to our hospital for treatment of a unilocular radiolucent lesion at the right anterior region of the maxilla. The lesion was excised. Microscopically, the tumor was composed of small nests or strands of odontogenic epithelial cells and amorphous eosinophilic globules of amyloid-like materials in a loose fibrous connective tissue stroma. The tumor epithelial cells were positive for pan-cytokeratins (AE1 and AE3). Langerhans cells demonstrated by anti-CD1a staining were found in nests or strands of tumor epithelial cells. The eosinophilic globules were positive for Congo red and showed green birefringence when subjected to polarized light. Review of the English literature revealed two cases of non-calcifying variant of intraosseous CEOT with Langerhans cells in the anterior and premolar regions of the maxilla. Taken together, we suggest that the non-calcifying, Langerhan cell-rich variant of CEOT may have a distinct predilection for occurrence in the anterior and premolar region of the maxilla in contrast to the classical CEOTs that usually occur in the molar and ascending ramus area of the mandible.     

Extraosseous calcifying epithelial odontogenic tumor (Pindborg tumor).

A 20-year-old man presented with a painless enlargement of the maxillary gingiva with no bone involvement. Microscopic examination with special stains confirmed the diagnosis of a clear cell variant of a calcifying epithelial odontogenic tumor (Pindborg tumor). This is the second reported case of such a variant in an extraosseous location.     

Ameloblastoma, calcifying epithelial odontogenic tumor, and glandular odontogenic cyst show a distinctive immunophenotype with some myoepithelial antigen expression

Background:  Odontogenic neoplasms have some morphologic overlap with salivary gland neoplasms, many of which show myoepithelial differentiation. In the 1980s, an ultrastructural study identified a population of myoepithelial-like cells in calcifying epithelial odontogenic tumor. Myoepithelial derived tumors have since been shown to have distinct immunohistochemical profiles.
Methods:  We examined a series of odontogenic neoplasms, including 11 ameloblastomas, four calcifying epithelial odontogenic tumors, five glandular odontogenic cysts (GOCs), and five keratocystic odontogenic tumors with a panel of myoepithelial-associated immunohistochemical stains. We also assessed representative control examples of oral mucosa, odontogenic rests, and dentigerous cysts.
Results:  All of the neoplastic and non-neoplastic oral epithelium-derived entities share a p63-positive, high molecular weight cytokeratin (CK5/6)-positive immunophenotype. Calponin reactivity was at least focally present in two of four calcifying epithelial odontogenic tumors, three of five GOCs, and 10 of 11 ameloblastomas; the sole completely non-reactive ameloblastoma represents a lung metastasis. One case of calcifying epithelial odontogenic tumor was focally positive for glial fibrillary acidic protein. However, other more definitive markers of myoepithelial differentiation, including S-100 and smooth muscle actin, were negative. Two of three calcifying epithelial odontogenic tumors and five of five GOCs were also positive for a low molecular weight cytokeratin (CK7).
Conclusions:  Ameloblastomas, GOCs, and calcifying epithelial odontogenic tumors show a distinctive immunophenotype which overlaps with that of myoepithelial-derived salivary gland neoplasms but does not provide definitive support for myoepithelial differentiation.     

Squamous odontogenic tumor. Review of the literature and case report

Squamous odontogenic tumor (SOT) is a rare, benign odontogenic tumor affecting both jaws in all age groups. The tumor is composed of squamous epithelial islands surrounded by a mature connective tissue stroma. This tumor may occasionally be misdiagnosed as ameloblastoma, squamous cell carcinoma, verrucous carcinoma and keratoacanthoma. A conservative surgical approach is the recommended treatment. An additional case is reported and the literature reviewed.     

Intramural calcifying epithelial odontogenic tumor

The calcifying epithelial odontogenic tumor (CEOT) is a rare lesion of the jaws. It accounts for about 1% of all odontogenic tumors. The CEOT occurs primarily in the molar-premolar region of the mandible, and 52% of cases are associated with an unerupted tooth. This report describes an unusual case in a 37-year-old woman. The tumor arose in the molar area of the right mandible, appeared radiographically as a radiolucent lesion, and was thought to be a dentigerous cyst in association with an impacted first molar. The lesion was enucleated. Microscopic examination showed it to be a dental sac, within which were the 3 elements of a typical CEOT: squamoid cells with eosinophilic cytoplasm, the homogeneous eosinophilic substance, and calcium salt deposits in the form of Liesegang rings.     

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提要：外周性牙源性肿瘤又称骨外型牙源性肿瘤或软组织牙源性肿瘤,主要发生在牙龈,包括牙源性真性肿瘤及错构瘤。外周性牙源性肿瘤约占所有牙源性肿瘤的4%左右,英文文献表明其中外周性牙源性纤维瘤最多见,其次是外周性成釉细胞瘤及外周性牙源性钙化囊性瘤。外周性牙源性肿瘤临床上容易与牙龈发生的炎症性或反应性病变相混淆,明确诊断依赖组织病理学检查。外周性牙源性肿瘤不包括骨内型牙源性肿瘤穿破骨皮质侵犯牙龈。外周性牙源性肿瘤预后普遍好于相应的骨内型肿瘤,但切除不彻底仍可复发,建议长期随访。     

Squamous odontogenic tumor: immunohistochemical identification of keratins

Two cases of squamous odontogenic tumors are described in terms of histopathology and keratin immunohistochemistry. Histopathologically, the lesions were composed of squamous epithelial islands without peripheral columnar cells and well-differentiated stromal tissue. Immunohistochemical detection of keratin proteins was done with the use of polyclonal antikeratin antiserum (TK, detecting 41 to 65 kDa keratins) and monoclonal antibodies (KL1, 55 to 57 kDa; PKK1, 40, 45, and 52.5 kDa). Staining for PKK1-detectable keratin was absent in tumor epithelial cells and that with KL1 and TK immunoreagents was confined to squamous cells, being strong in the keratinized cells. In view of the results, the squamous odontogenic tumor appears to arise from the rests of Malassez in periodontal tissue rather than from oral squamous epithelium.     

Peripheral clear cell calcifying epithelial odontogenic tumor. Report of a case

Calcifying epithelial odontogenic tumor (CEOT) is an uncommon, locally aggressive odontogenic tumor representing less than 1% of all odontogenic tumors. Clear cells have been described in CEOT, ameloblastomas, calcifying odontogenic cysts, lateral periodontal, and gingival cysts. It is not yet clear if the clear cell variants of the odontogenic tumors have a different biologic behavior, even if it seems that these tumors are more aggressive with an higher recurrence rate. The authors present a case of a peripheral clear cell CEOT (CCCEOT). It is a rare lesion with only 9 cases reported in the literature. The lesion was removed by excision and no recurrence was found after a 4-year follow-up.