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

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.

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Journal
Archiv für Pathologische Anatomie und Physiologie und für Klinische Medicin
Published
2025-02-22
DOI
https://doi.org/10.1007/s00428-025-04053-1
Citations
1
Primary Topic
Head and Neck Surgical Oncology
Type
article
Field-Weighted Citation Impact
2.01

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

Stephan Ihrler, Radek Šíma, Cécile Badoual, Ilmo Leivo et al.
1 citations
Archiv für Pathologische Anatomie und Physiologie und für Klinische Medicin
Head and Neck Surgical Oncology
2.01
article

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

Stephan Ihrler, Radek Šíma, Cécile Badoual, Ilmo Leivo, Ján Laco, Abbas Agaimy, Niels J. Rupp, Sarina K. Mueller, Sami Ventelä, Carlos E. Bacchi, Petr Šteiner, Ivan Damjanov, Martina Bradová, Petr Martínek, Tomáš Vaněček, Éva Compérat, Alena Skálová, Stanislav Kormunda Ing, Michal Michal
article en
1 citations

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.

Archiv für Pathologische Anatomie und Physiologie und für Klinische MedicinVol. 486(6)
University of Kansas (US), University of Turku (FI), Friedrich-Alexander-Universität Erlangen-Nürnberg (DE), Université Paris Cité (FR), Charles University (CZ), Turku University Hospital (FI), Sorbonne Université (FR), Universitätsklinikum Erlangen (DE), Laboratório Bacchi (BR), Assistance Publique – Hôpitaux de Paris (FR), University Hospital of Zurich (CH), Hôpital Européen Georges-Pompidou (FR), Biopticka Laborator (Czechia) (CZ), Dermatopathologie Friedrichshafen (DE), University Hospital Hradec Králové (CZ), Comprehensive Cancer Center Erlangen (DE), Hôpital Tenon (FR), Medical University of Vienna (AT)
Univerzita Karlova v Praze
Good health and well-being
Openalex Percentile: Top 19%
Head and Neck Surgical Oncology
2.01
DOIOpen Access PDFOpenAlex
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