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.     

Combined calcifying epithelial odontogenic tumor and adenomatoid odontogenic tumor

A case of combined epithelial odontogenic tumor associated with an unerupted maxillary canine tooth is described. The relative proportion of adenomatoid odontogenic tumor tissue and calcifying epithelial odontogenic tumor areas in a given tumor in determining the behaviour and growth potential of this entity is discussed.     

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。     

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.     

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.     

Peripheral calcifying epithelial odontogenic tumor

Most of the calcifying epithelial odontogenic tumors (CEOT) are central lesions of the jaws, but the CEOT occurs infrequently on the gingiva. A cases of peripheral CEOT arising in the right upper molar gingiva of a 31-year-old Japanese woman is presented. A direct communication between the overlying gingival epithelium and the tumor was found by examination of serial sections. As a result, it was thought that one of the sources of peripheral CEOT might be gingival epithelium with potential proliferative activity.     

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

Calcifying epithelial odontogenic tumor (CEOT) is a benign, locally aggressive odontogenic neoplasm characterized by sheets and nests of epithelial cells with deeply eosinophilic or occasionally clear cytoplasm, calcifications, and eosinophilic amorphous material that stains positive for amyloid. Although many cases of CEOT are associated with impacted teeth and occasionally appear radiographically as dentigerous cysts, a true cystic variant has not been previously reported. We report a 15-year-old white male with a large cystic maxillary lesion that filled most of the left maxillary sinus. It deformed the medial wall, the inferior orbital floor, and caused narrowing of the left inferior meatus. Histologically, the cystic lining showed characteristics of CEOT. An intraluminal component that featured histologic characteristics of CEOT was identified during surgery. The lesion was enucleated and the postsurgical course of the patient was uneventful. Because follow-up has been for less than 1 year, a meaningful long-term prognosis cannot be determined at present. However, the patient has not reported any symptoms or signs of recurrence during the follow-up period.     

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.     

钙化上皮瘤及牙源性钙化上皮瘤的临床病理分析

目的：探讨钙化上皮瘤及牙源性钙化上皮瘤的临床表现、诊断及外科处理原则。方法：对13例钙化上皮瘤和4例牙源性钙化上皮瘤患者的临床资料进行回顾性分析。结果：所有患者均经手术治疗。钙化上皮瘤患者平均年龄11．2岁,小于10岁者占69．2％（10例）;位于头颈部8例（61．5％）,四肢4例（30．8％）,躯干1例（7．7％）。牙源性钙化上皮瘤患者平均年龄38．75岁,均位于颌骨。两种肿瘤无明显性别差异,均有复发、恶变的报道。结论：钙化上皮瘤和牙源性钙化上皮瘤在临床表现和病理诊断上具有明显差别,是两种不同的肿瘤;牙源性钙化上皮瘤具有局部浸润性生长,手术切除必须彻底。     

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.     

Ghost cell odontogenic carcinoma arising in the background of a benign calcifying cystic odontogenic tumor of the mandible

Ghost cell odontogenic carcinoma (GCOC) is a rare malignant variant of odontogenic tumor with ghost cells; only 29 cases are documented. Our patient was a 68-year-old man with a painless, well-defined, radiolucent swelling of the mandibular gingiva in the right incisor-to-molar region. It was diagnosed as a benign calcifying cystic odontogenic tumor (CCOT) on fenestration biopsy. Eighteen years later, he returned with swelling in the same area. The lesion was excised, diagnosed as GCOC, and considered a secondary malignant manifestation of the benign CCOT. No adjuvant chemotherapy or radiotherapy was administered, and his postoperative course was uneventful for 48 months, with no recurrence or distant metastasis. Among the 30 reported cases of GCOC, the mean age at diagnosis was 40.3 years, 22 (73%) involved the maxilla. Twelve (40%) were secondary malignant manifestations of benign CCOTs or dentinogenic ghost cell tumors. Five patients died of recurrence or distant metastasis.     

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.     

A variant of calcifying epithelial odontogenic tumor with Langerhans cells

A variant of calcifying epithelial odontogenic tumor (CEOT) with Langerhans cells is reported. Compared to a typical CEOT, the tumor islands of this case were thin and composed of a small number of polyhedral epithelial cells. Almost no calcification of homogeneous eosinophilic materials was observed. In addition, clear cells which structurally corresponded to Langerhans cell were intermingled in the epithelial islands. These cells stain positively for S-100 protein, lysozome, MT 1, LN-3 and OKT 6 antibodies, but not for keratin antibody. Electronmicroscopic examination revealed the rod-shaped and racket-shaped structures called Birbeck's granules in the cytoplasm of these clear cells. Our observations indicate a variant case of CEOT with Langerhans cells in tumor nests.     

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.     

骨外型牙源性钙化上皮瘤1例报告

牙源性钙化上皮瘤是一种良性肿瘤，也称Pindborg瘤，可分为骨内型和骨外型2种，主要发生于颌骨内，偶见于颌骨外。本文报告1例发生于口底的牙源性钙化上皮瘤，其临床症状与口底皮样囊肿相似．术后经组织病理学确诊，主要组织病理学特征是嗜酸性环状钙化团块。因其具有局部侵袭性，为避免复发，提倡完整切除肿物。