High-grade transformation of a polymorphous adenocarcinoma of the salivary gland: a case report and review of the literature

Front Oncol. 2023 Sep 18:13:1245043. doi: 10.3389/fonc.2023.1245043. eCollection 2023.

Abstract

Background: Polymorphous adenocarcinoma (PAC) represents the second most widespread neoplasm of the minor salivary glands. These tumors rarely develop a histological progression from low-grade to high-grade malignancy, named "high-grade transformation" (HGT). Only nine cases are described in literature.

Case description: Here, we describe the case of a 76-year-old male patient with a PAC recurrence of the oral floor displaying HGT, and we explore the tumor cytomorphological features, genomic profiling, and the patient's clinical management. The tumor mass was characterized by poorly atypical cellular elements with vesicular nuclei and comedonecrosis foci. The growth pattern was predominantly solid, tubular, and cribriform. The lesion did not show microsatellite instability or targeted molecular alterations. The case was successfully treated with radical surgery followed by radiotherapy.

Conclusion: We report for the first time the recurrence of a PAC with HGT arising in the oral floor after 20 years from the primary lesion. These preliminary data and the literature analysis enhance the knowledge of this extremely rare disease.

Keywords: gene expression profiling; high-grade transformation; minor salivary gland; oral floor; polymorphous adenocarcinoma.

Publication types

  • Case Reports

Grants and funding

This work was partly supported by the contribution of Ricerca Corrente by the Italian Ministry of Health within the research line Precision, gender and ethnicity-based medicine and geroscience: genetic-molecular mechanisms in the development, characterization and treatment of tumors.