Calcifying odontogenic cyst: Report of 2 cases and review of literature

Highlights

  • Calcifying odontogenic cyst highlights:

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      Calcifying odontogenic cyst (COC) is an uncommon odontogenic cyst known to affect a broad age range.

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      In 20% of cases, it occurs in association with an odontoma.

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      Recurrence rate and malignant transformation are not clearly documented, some studies suggested to be <1%

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      Here we are presenting 2 cases of COC with similar histopathology abd interesting clinical appearance.

Introduction

Calcifying odontogenic cyst (COC) was first reported by Gorlin in 1962 [ ], and Gold in 1963 [ ]. It accounts for 0.3–0.8% of all odontogenic tumors [ ]. COC is a developmental odontogenic lesion which arises from dental lamina rests (rests of Serres) within bone or soft tissue [ ]. COC. This cyst can present as intraosseous (central) lesion or gingival (peripheral) lesion [ ]. In 20% of cases, it occurs in association with an odontoma [ , ]. Depending on the COC variant or behavior, treatment has been suggested as (1) enucleation and curettage [ ], (2)tube decompression followed by enucleation and curettage [ ], (3) enucleation, curettage and peripheral ostectomy, or (4) segmental resection (for lesions with neoplastic characteristics) [ ].

The purpose of this article is to present two examples of COC with a remarkably similar presentation and surgical intervention. Associated review of literature will also be included.

Patients and methods

This was a case series of two patients with COC treated by Oral and Maxillofacial Surgery at Emory University. Institutional review board exemption was obtained. Patients were evaluated by history, clinical examination, and imaging studies (panoramic radiograph and computed tomography scan, CT). Both lesions were submitted for and histopathological evaluation. Patients were followed for at least 1 year.

Case 1

A 27-year-old male reported a two-year history of painless swelling of maxilla. Physical examination revealed extensive left facial swelling from medial canthus to upper lip causing distortion of nares and ala, obstructing left nasolabial fold, and causing left-sided lip incompetence ( Fig. 1 A and B). Intraoral examination revealed left-sided fluctuant vestibular swelling extending from the right maxillary lateral incisor to the left maxillary second molar ( Fig. 1 C). The lesion caused a step in occlusion between right and left central incisors, a left-sided premature posterior contact, and an anterior open bite. There was a minimum palatal expansion. The overlying mucosa was normal in color and texture.

Fig. 1
Frontal (A), profile (B), intraoral views (C), panoramic radiograph (D), and axial (E), sagittal (F) and coronal (G) views of CT scan, intraoperative picture (H) of 27-year-old male who presented with left facial swelling. Lesion measured 7.8 × 4.8 × 4.6 cm and had a radiopaque structure with a comparable density to tooth enamel in center of lesion (arrow). Biopsy confirmed calcifying odontogenic cyst.

Panoramic radiograph ( Fig. 1 D) and CT ( Fig. 1 E–G) revealed a well-defined, unilocular, expansile, radiolucent lesion with a thin sclerotic rim measuring 7.8 × 4.8 × 4.6 cm and encompassing majority of left maxillary sinus and extending from right maxillary central incisor to left maxillary third molar. Lesion had a central radiopaque structure with density comparable to tooth enamel. All associated teeth had apical root resorption. Incisional biopsy of the lesion was performed under local anesthesia. Histopathologic evaluation was consistent with calcifying odontogenic cystic. The patient underwent enucleation and curettage of lesion using an intraoral approach under general anesthesia. Endodontic treatment of associated teeth was recommended. At one year follow up, the lesion did not recur.

Case 2

A 17-year-old female presented with an incidental finding of lesion of left maxilla. Physical examination revealed no facial asymmetries ( Fig. 2 A and B). Intraorally, there was subtle left maxillary vestibule expansion from canine to second molar ( Fig. 2 C). Panoramic radiograph ( Fig. 2 D) and CT ( Fig. 2 E–G) revealed a well-defined, unilocular, radiolucent lesion of left maxilla measuring 4.1 × 2.3 × 4.2 cm, extending from left maxillary central incisor to left maxillary third molar, expanding into maxillary sinus, and obliterating the left maxillary outflow tract. An amorphous radiopaque density was visualized within the lesion. All associated teeth had severe apical root resorption. The patient underwent enucleation and curettage of lesion using an intraoral approach under general anesthesia. Endodontic treatment of associated teeth was recommended. At two year follow up, the lesion did not recur.

Aug 8, 2020 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Calcifying odontogenic cyst: Report of 2 cases and review of literature
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