Assessment of biologically aggressive, recurrent glandular odontogenic cysts for mastermind-like 2 (MAML2) rearrangements: histopathologic and fluorescent in situ hybridization (FISH) Findings in eleven cases

Abstract

Background

Glandular odontogenic cyst (GOC) demonstrates a significant predilection toward localized biologic aggressiveness and recurrence. GOC shares certain histopathologic features with intraosseous mucoepidermoid carcinoma (IMEC). The current investigation evaluates a group of recurrent, biologically aggressive GOCs to determine if any cases demonstrated unique histologic features or Mastermind – like2 (MAML2) rearrangements common to IMEC.

Methods

Microscopic slides from eleven previously diagnosed GOGs were stained with hematoxylin and eosin and assessed by two study participants for ten classic histopathologic features required to establish a diagnosis of GOC. Cases were evaluated utilizing break-apart fluorescent in situ hybridization (FISH) analysis for the presence of MAML2 gene rearrangements. Clinical and demographic data on all patients were recorded.

Results

The mean age for patients included in the study was 55.27 years with a range of 36 to 72 years. The most common presenting symptom was a jaw expansion and all cysts presented initially as a unilocular or multilocular radiolucency. CYSTS displayed a minimum of 6 of 10 histologic parameters necessary for a diagnosis of GOC. One case demonstrated MAML2 rearrangements by FISH. That case also showed marked ciliation of cyst lining epithelial cells and extensive mucous secreting goblet cell proliferation.

Conclusion

Findings in the current study are in concert with previous investigations, and although this study finds ONLY LIMITED molecular evidence to support the premise that recurrent biologically aggressive GOCs are a precursor to IMEC, detection of MAML2 rearrangements in one case suggests that such a theoretic transition, while rare, is possible.

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