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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?

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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.

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The data including clinical information, radiograph, and histopathologic preparations are available upon request.

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Funding

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.

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Authors and Affiliations

  1. Division of Oral and Maxillofacial Pathology, School of Dentistry, University of Minnesota, 515 Delaware Street SE, 16-116B, Minneapolis, MN, 55455, USA

    Ioannis G. Koutlas

  2. Faculty of Dental Medicine, Université de Montréal, Montréal, Québec, H3T 1J4, Canada

    Katia Julissa Ponce, Rima-Marie Wazen & Antonio Nanci

  3. Department of Biochemistry and Molecular Medicine, Faculty of Medicine, Université de Montréal, Montréal, Québec, H3T 1J4, Canada

    Antonio Nanci

Authors
  1. Ioannis G. Koutlas
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  2. Katia Julissa Ponce
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  4. Antonio Nanci
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Contributions

IGK designed the study, analyzed the data, authored the paper and is the corresponding author. KJP, RWM and AN provided immunohistochemical stains and commentary for ODAM, AMTN and SCPPPQ1 which were performed at the University of Montreal. The paper was critically reviewed and approved by all authors.

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Correspondence to Ioannis G. Koutlas.

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The authors declare no conflicts of interest.

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The study was performed in accordance with the 1964 Helsinki Declaration standards.

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The study was exempt from the University of Minnesota Institutional Review Board.

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Koutlas, I.G., Ponce, K.J., Wazen, RM. et al. 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?. Head and Neck Pathol 16, 587–595 (2022). https://doi.org/10.1007/s12105-021-01369-7

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