Robert O Greer1,2,3, Jeffrey Eskendri4, Paul Freedman4,5, Moni Ahmadian4, Aline Murakami-Walter3, Marileila Varella-Garcia3. 1. Division of Oral and Maxillofacial Pathology, School of Dental Medicine, Anschutz Medical Campus, University of Colorado, Aurora, CO, USA. 2. Department of Pathology, School of Medicine, Anschutz Medical Campus, University of Colorado, Aurora, CO, USA. 3. Division of Medical Oncology and University of Colorado Cancer Center, Anschutz Medical Campus, University of Colorado, Aurora, CO, USA. 4. Section of Oral and Maxillofacial Pathology, New York Presbyterian/Queens, Flushing, NY, USA. 5. Oral Pathology Laboratory Inc., Flushing, NY, USA.
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 whether any cases demonstrated unique histologic features or mastermind-like2 (MAML2) rearrangements common to IMEC. METHODS: Microscopic slides from 11 previously diagnosed GOCs were stained with hematoxylin and eosin and assessed by 2 study participants for 10 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 1 case suggests that such a theoretic transition, while rare, is possible.
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 whether any cases demonstrated unique histologic features or mastermind-like2 (MAML2) rearrangements common to IMEC. METHODS: Microscopic slides from 11 previously diagnosed GOCs were stained with hematoxylin and eosin and assessed by 2 study participants for 10 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 1 case suggests that such a theoretic transition, while rare, is possible.
Authors: Peter P Luk; James Wykes; Christina I Selinger; Rafael Ekmejian; Jessica Tay; Kan Gao; Timothy J Eviston; Trina Lum; Sandra A O'Toole; Jonathan R Clark; Ruta Gupta Journal: Oral Surg Oral Med Oral Pathol Oral Radiol Date: 2016-01-09
Authors: Marco Mascitti; Andrea Santarelli; Antonio Sabatucci; Maurizio Procaccini; Lorenzo Lo Muzio; Antonio Zizzi; Corrado Rubini Journal: Open Dent J Date: 2014-02-07