Sinonasal adenoid cystic carcinomas accompanied by seromucinous hamartoma and/or atypical sinonasal glands arising from seromucinous hamartoma: insight into their histogenesis

Bradová M, Agaimy A, Laco J, Martínek P, Ing SK, Badoual C, Damjanov I, Leivo I, Bacchi CE, Comperat E, Ihrler S, Rupp NJ, Šíma R, Šteiner P, Vaněček T, Müller SK, Ventelä S, Skálová A, Michal M (2025)

Publication Type: Journal article

Publication year: 2025

Journal

Virchows Archiv Springer Verlag (Germany)

DOI: 10.1007/s00428-025-04053-1

Abstract

The pathology of reactive, dysplastic, and neoplastic sinonasal seromucinous glands is complex, and their contribution to tumorigenesis of sinonasal carcinomas remains controversial. In our practice, we have observed the presence of respiratory epithelial adenomatoid hamartomas (REAH) and seromucinous hamartomas (SH) associated with adenoid cystic carcinomas (AdCC) in a subset of cases. In many of these cases, genuine atypical features and dysplastic characteristics of the glands were noted at the interface of SH and AdCC. To investigate this phenomenon further, 88 sinonasal AdCC cases were selected from the authors’ files and analyzed histologically, immunohistochemically, and genetically searching for MYB/MYBL1 and NFIB gene fusions. HPV testing was also performed. Univariate statistical analysis was conducted on our cohort. Thirty-one cases (35%) showed features of atypical sinonasal glands arising in SH (ASGSH) at the SH-AdCC interface, characterized by bilayered epithelium, architectural disarray, mild nuclear polymorphism, and atypia, sometimes with colloid-like material in the lumen. The MYB immunomarker was negative in 14 ASGSHs (with a positive internal control in AdCC cells), while only two cases showed faint and moderate to weak expression of the antibody in ASGSH glands. In 12 cases, the immunostaining of ASGSH could not be properly assessed, while AdCC cells were negative. The immunostaining was not performed in five cases. Our findings suggest that a subset of sinonasal AdCC may originate in a multistep dysplastic process within SH, consistent with an SH-ASGSH-AdCC progression sequence.

Authors with CRIS profile

Abbas Agaimy Lehrstuhl für Allgemeine Pathologie und Pathologische Anatomie Sarina Katrin Ursel Müller-Hübner Department of Otorhinolaryngology – Head and Neck Surgery (HNO-Klinik)

Involved external institutions

Univerzita Karlova v Praze / Charles University in Prague CZ Czech Republic (CZ) University Hospital Hradec Králové / Fakultní Nemocnice Hradec Králové CZ Czech Republic (CZ) Georges Pompidou European Hospital / Hôpital Européen Georges-Pompidou (HEGP) FR France (FR) University of Kansas Medical Center US United States (USA) (US) Turku University Hospital / Turun yliopistollinen keskussairaala (TYKS) FI Finland (FI) Laboratório Bacchi Consultoria em Patologia BR Brazil (BR) Hôpital Tenon FR France (FR) Universitätsspital Zürich (USZ) CH Switzerland (CH)

How to cite

APA:

Bradová, M., Agaimy, A., Laco, J., Martínek, P., Ing, S.K., Badoual, C.,... Michal, M. (2025). Sinonasal adenoid cystic carcinomas accompanied by seromucinous hamartoma and/or atypical sinonasal glands arising from seromucinous hamartoma: insight into their histogenesis. Virchows Archiv. https://doi.org/10.1007/s00428-025-04053-1

MLA:

Bradová, Martina, et al. "Sinonasal adenoid cystic carcinomas accompanied by seromucinous hamartoma and/or atypical sinonasal glands arising from seromucinous hamartoma: insight into their histogenesis." Virchows Archiv (2025).

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