An Unusual Maxillary Tumor with Tubuloductal Epithelial Structures, Solid Epithelial Nests and Stromal Odontogenic Ameloblast-Associated Protein Deposits. Tubuloductal/Syringoid Variant of Central Odontogenic Fibroma with Amyloid?

Ioannis G. Koutlas, Katia Julissa Ponce, Rima Marie Wazen, Antonio Nanci

Research output: Contribution to journalArticlepeer-review

Abstract

Glandular tumors of jaw bones present, most often, histopathologic features of salivary gland and, rarely, of cutaneous glandular neoplasms. They are thought to originate from odontogenic epithelium. An unusual maxillary tumor presenting as a radiolucency in the periapical area of the right permanent lateral incisor of a 74-year-old male is presented causing root resorption. Preparations revealed occasionally branching tubular cords and ductal structures characterized, mostly, by a bilayer composed of luminal cuboidal to low columnar cytokeratin (CK) 7, Ber-EP4 and occasionally CK8/18 positive cells, and abluminal, CK5/6 positive, basal/basaloid cells revealing nuclear reactivity for p63/p40. Smooth muscle actin and calponin were negative, save for a single focus of calponin positive cells, confirming absence of myoepithelial support or epithelial mesenchymal transition. CK19 exhibited staining of both layers, the luminal being more intense. Eosinophilic secretory material and, occasionally, a luminal pellicle were decorated with CK8/18 and polyclonal carcinoembryonic antigen (CEA). CD1a identified only rare Langerhans’ cells and Ki67 decorated 1–2% of abluminal cell nuclei. Small solid nests of epithelial cells were also present. Infrequently, an apparent transition of a nest into a tubular structure was appreciated. The partially inflamed stroma featured multiple hyalinized acellular deposits consistent with amyloid, as confirmed by bright orange Congo red reactivity with apple-green birefringence, which reacted with odontogenic ameloblast-associated (ODAM) protein antibody but not with antibodies for amelotin and secretory calcium-binding phosphoprotein proline-glutamine rich 1. Based on the above, the diagnosis of tubuloductal/syringoid variant of central odontogenic fibroma with ODAM amyloid is favored.

Original languageEnglish (US)
JournalHead and Neck Pathology
Early online dateAug 2 2021
DOIs
StatePublished - Aug 2 2021

Bibliographical note

Funding Information:
The study was partially supported by the Canadian Institutes of Health Research [Antonio Nucci (CIHR MOP-110972)], and the Network for Oral and Bone Health Research (FRQ-S). Antonio Nucci is recipient of a Canada Research Chair in Calcified Tissues, Biomaterials and Structural Imaging.

Publisher Copyright:
© 2021, The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.

Keywords

  • Amyloid
  • Central odontogenic fibroma
  • Odontogenic ameloblast-associated protein
  • Tubular adenoma

PubMed: MeSH publication types

  • Journal Article

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