Glandular Odontogenic Cyst Excision with Immediate Reconstruction: A Case Report
First reports of this cyst used the term sialo-odontogenic cyst, based on the specimen’s microscopic similar glandular tissue. The World Health Organization (WHO) originally classified the GOC as a developmental odontogenic epithelial cyst under the titles glandular odontogenic cyst or sialo-odontogenic cyst. At that time the WHO believed there to be no evidence for the GOC to be of salivary gland origin. In recent years, the issue of the origin of the GOC seems to be shifting towards support of odontogenic origin rather than sialogenic origin. Several cases of “hybrid” lesions of GOC with other odontogenic tumors are an indicator of odontogenic origin. Glandular Odontogenic cysts have also shown to possess minimal or completely lack expression of markers that do not support a sialogenic origin such as, epithelial membrane antigen (EMA) and mammary serine protease inhibitor (MASPIN).
Correct diagnosis is of significant clinical importance, since the GOC has an aggressive potential, a high incidence of cortical perforation and a relatively high rate of recurrence. Definitive diagnosis can be challenging due to similarities in histopathologic features between the GOC and lesions such as dentigerous cysts with mucous metaplasia, botryoid cyst, and most importantly low-grade mucoepidermoid carcinoma (MEPCa).
A 30 year old Caucasian male presented to our Oral & Maxillofacial surgery clinic with history of a rapidly expanding lesion of the anterior mandible. The lesion extended from the area of teeth #’s 21-28. Superiorly the lesion occupied the alveolar ridge and extended inferiorly to within 2.5cm of the inferior border of the mandible. The lingual cortex of the anterior mandible was intact but the lesion had completely eroded through the buccal cortex. An incisional biopsy was performed and signed out as a glandular Odontogenic cyst. The patient was taken to the operating room for curettage coupled with peripheral ostectomy. To establish an intact border for the specimen, removal of the lesion included the buccal cortex periosteum covering. The lesion was approximately 1.5cm x 3.5cm x2.0 cm, soft, friable and non-encapsulated. Immediate reconstruction of the defect was performed with cancellous bone harvested from the anterior iliac crest. Alloderm was laid over the bone graft to augment soft tissue closure. Close follow up at one week, two weeks, one month, three months, and six months have clinically and radiographically demonstrated no recurrence to date.
Immediate reconstruction versus delayed reconstruction protocols have not yet been established in the literature. We decided on immediate reconstruction with our patient using several factors. Our patient was in good systemic health, was young in age, the patient was not expected to have any difficulty maintaining a healthy caloric diet and the patient requested immediate reconstruction. Curettage with immediate reconstruction may be a viable option in the treatment of glandular odontogenic cysts if certain criteria are met. These criteria may include absence of systemic disease, young age, and possibility of performing optimum surgical resection with appropriate margins while maintaining mandibular continuity. Further studies are needed to establish definitive surgical protocols in the treatment of glandular odontogenic cysts.
Kaplan I et al (2005a). Glandular odontogenic cyst: treatment and recurrence. J Oral Maxillofac Surg63: 435–441.
Fowler CB et al (2011) Glandular odontogenic cyst: analysis of 46 cases with special emphasis on microscopic criteria for diagnosis. Head Neck Pathol 5:364–375