Recurrent glandular odontogenic cysts: a series of seven cases

Introduction

We present a case series of recurrent glandular odontogenic cysts (rGOC), a rare, aggressive odontogenic cyst seen commonly in the anterior mandible of middle-aged women with reported recurrence rates of 22%. Our objective is to analyze the clinical and histological features of rGOCs from our institution.

Materials and Methods

The University of Pittsburgh Pathology archives were searched for rGOCs (1993-2022). A total of 265 glandular odontogenic cysts were diagnosed with a recurrence rate of 2.3%. We reviewed the slides and pathology reports for the clinicopathologic features of our recurrent cases and paired primary cases, when available.

Results

We assembled a series of six patients with seven rGOCs, with one patient having two recurrences. Average time to recurrence was 10.7 years (range: 2.3-22.6 years). With a mean age at initial diagnosis of 50.3 years (range: 24-69) and no sex predilection (M:F 1:1). All cases presented in the anterior gnathic region with an even jaw distribution (maxilla:mandible 1:1). Four tumors in two patients were MAML2 translocation tested and negative, including one patient with two recurrences. Histologically, rGOCs had minimal inflammation and a flat epithelial connective tissue interface; separation at the epithelial-connective was noted in all cases, at least focally. Another interesting finding in the rGOCs was subepithelial hyalinization (epithelial inductive effect). All original and recurrent tumors reviewed demonstrated variable thickness, clear cells, and multiple compartments. In cases where both the original slides and recurrence were available for review, the recurrence lacked/lost on average 3.3 features. Microcysts were present in all primary cysts reviewed, but not seen in recurrent lesions.

Conclusions

In our single institution experience, rGOC is most common in the anterior gnathic region with lower recurrence rates than stated in the literature. Variable surgical management strategies are a confounding factor. Recurrent lesions histologically contained fewer characteristic GOC features than initial lesions.