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.

Bioptic Laboratory Ltd Plzen Czech Republic

Consultoria Em Patologia Botucatu São Paulo Brazil

Department of Otorhinolaryngology and Head and Neck Surgery University Hospital Erlangen Erlangen Germany

Department of Otorhinolaryngology Turku University Hospital Turku Finland

Department of Pathology and Laboratory Medicine The University of Kansas School of Medicine Kansas City KS USA

Department of Pathology and Molecular Pathology University Hospital Zurich Zurich Switzerland

Department of Pathology Medical University of Vienna Vienna Austria

Department of Pathology Tenon Hospital Sorbonne University Paris France

Department of Pathology Turku University Hospital Turku Finland

Dermpath Muenchen Germany

Institute of Biomedicine Pathology University of Turku Turku Finland

Institute of Pathology University Hospital Erlangen Friedrich Alexander University Erlangen Nürnberg Erlangen EMN Erlangen Germany

Molecular and Genetic Laboratory Bioptic Laboratory Ltd Plzen Czech Republic

Service d'Anatomo Pathologie Department of Pathology Hôpital Européen G Pompidou APHP Université de Paris 20 40 Rue Leblanc 75015 Paris France

Sikl's Department of Pathology Charles University Faculty of Medicine in Plzen E Benese 13 305 99 Pilsen Czech Republic

The Fingerland Department of Pathology Charles University Faculty of Medicine and University Hospital Hradec Kralove Czechia Czech Republic

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