Sequencing of Sclerosing Microcystic Adenocarcinoma Identifies Mutational Burden and Somatic Variants Associated With Tumorigenesis

Anticancer Res. 2020 Nov;40(11):6375-6379. doi: 10.21873/anticanres.14658.

Abstract

Background/aim: Sclerosing microcystic adenocarcinoma (SMA) is a rare oral cavity neoplasia, histologically resembling microcystic adnexal carcinoma (MAC) of the skin. Only nine SMA cases have been reported in the literature, frequently in the context of immunosuppression; SMA has not been recognized in the most recent WHO tumor classification. We sought to identify potential molecular mechanisms of tumorigenesis in a case of SMA relative to those known for MAC.

Case report: A 41-year-old female with psoriatic arthritis undergoing immunosuppression therapy presented with a tongue mass. Biopsy revealed a diagnosis of SMA. Partial glossectomy and neck dissection showed no residual tumor or nodal disease.

Results: whole exome sequencing revealed moderate mutational burden and putative loss of function mutations in CDK11B but no overlap with known MAC mutations.

Conclusion: We characterized the genomic profile of SMA for the first time, identifying both mutational burden and unique somatic variants associated with tumorigenesis.

Keywords: Next-generation sequencing; immunosuppression; oral cancer; sclerosing microcystic adenocarcinoma.

Publication types

  • Case Reports

MeSH terms

  • Adenocarcinoma / genetics*
  • Adenocarcinoma / pathology
  • Adenoma / genetics*
  • Adenoma / pathology
  • Adult
  • Carcinogenesis / genetics
  • Cyclin-Dependent Kinases / genetics*
  • Exome Sequencing
  • Female
  • Gene Expression Regulation, Neoplastic / genetics
  • Humans
  • Mouth / metabolism
  • Mouth / pathology
  • Mutation / genetics
  • Neoplasms / genetics
  • Neoplasms / pathology

Substances

  • CDK11B protein, human
  • Cyclin-Dependent Kinases