Cystic Variant of Calcifying Epithelial Odontogenic Tumor

Abstract

To the Editor: The calcifying epithelial odontogenic tumor (CEOT) is an uncommon benign tumor of epithelial origin, representing <1% of all odontogenic tumors. Among the histological variants of CEOT, the true cystic variant of CEOT is extremely rare, with only 3 welldocumented cases reported in the English-language literature. [1][2][3] In this report, we describe the clinical, radiographic, and histopathological features of a case of cystic variant of CEOT.A 22-year-old male patient was referred to evaluation of a gingival swelling with approximately 8 months of evolution. Intraoral examination revealed a painless, dome-shaped, nodular lesion, which presented a bluish color and soft/cystic consistency to palpation. The nodule was located on the attached gingival mucosa and mobile alveolar mucosa, between the right mandibular canine and right mandibular first premolar (Fig. 1A). Radiographically, a well-circumscribed, ovoid, unilocular, mixed radiopaque, and radiolucent lesion was observed, measuring 2.0 cm of extension. The lesion was located between the right mandibular canine and first premolar and caused divergence of the roots of the involved teeth. Moreover, discrete root resorption on the first premolar was also observed (Fig. 1B). Occlusal radiography showed expansion and rupture of the buccal cortical bone, with the presence of numerous small, round, and radiopaque formations. An exploratory needle aspiration was performed, which showed an abundant amount of liquid. After aspiration, a significant decrease in swelling was observed. According to the clinical and radiographic findings, a calcifying cystic odontogenic tumor was the main hypothesis of diagnosis.Under local anesthesia, the lesion was excised, with subsequent curettage of the surrounding bone tissue (Fig. 1C). Microscopically, most of the lesion appeared as cystic cavities lined by polyhedral epithelial cells associated with numerous calcifications (Fig. 1D). In the cystic lining, these polyhedral epithelial cells were interspersed by calcifications (Fig. 1E). Extensive concentric calcifications were frequent in the stroma and between the tumor cells. Clear cells were commonly observed. In addition, focal areas with amorphous, eosinophilic, and hyalinized material positive for Congo red staining were also noticed (Fig. 1F). Thus, the diagnosis of calcifying epithelial odontogenic tumor, cystic variant, was established. One year after the excision, there was complete bone healing. Currently, 10 years after treatment, no clinical and radiographic signs of recurrence were observed.The most common clinical feature of CEOT is a firm, painless expansive mass located in the posterior region of the mandible. 4 In contrast, the current case showed an unusual presentation, appearing as a well-circumscribed gingival nodule with a blue-colored surface and soft consistency. These atypical features may be related to cystic presentation of the CEOT, which showed an expansive growth rather than infiltrative. In the same way, radiographically, this case show...

CEOT Variants or Entities: Time for a Rethink? A Case Series with Review of the Literature

“…The lesion was enucleated. A number of similar cases have been reported [48][49][50], and subsequently, a microcystic variant has also been described [25]. In this lesion, a pseudo-glandular appearance was reported in association with otherwise rather conventional CEOT histology.…”

Microcystic Calcifying Epithelial Odontogenic Tumor

“…Interestingly, all cystic CEOT cases showed gross expansion of cortical bone, average size was 3.3 cm in the greatest diameter. To the best of our knowledge, our case is the second microcystic variant of CEOT reported [13].…”

“…In 2006, Gopalakrishnan et al reported the first case of cystic variant of CEOT in a 15-year-old male with an expansive mixed lesion of the posterior maxilla [10]. Since then, only three additional CEOT cases showing considerable cystic or microcystic pattern have been reported [11][12][13]. All patients were males with a median age of 24.5 years (ranging from 15 to 31).…”

“…Two cases were located in the posterior maxilla, related to an unerupted displaced tooth, and showing maxillary sinus extension [10,11], which is also a common finding in conventional maxillary CEOTs [5]. Other two cases were located in the toothbearing regions of the body of the mandible with root displacement or resorption, one of them extending posteriorly into the ramus [12,13]. Interestingly, all cystic CEOT cases showed gross expansion of cortical bone, average size was 3.3 cm in the greatest diameter.…”

“…Often, CEOT-like epithelial nests may be observed in dental follicles, and its significance even not completely understood, are not considered early stages of CEOT [1,2,7]. Although most cases are solid tumors, rare predominantly cystic CEOT cases have been reported (Table 1) [10][11][12][13][14]. We herein present a well-documented case of microcystic CEOT located in the posterior mandible of a 42-year-old female.…”