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Chronic Sclerosing Sialadenitis of the Submandibular Gland and its Histopathological Spectrum in the IgG4-Related Disease: a Series of 17 Cases

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Abstract

Purpose

This study aimed to characterize the histopathological immunohistochemical features of chronic sclerosing sialadenitis, emphasizing the IgG4-related disease.

Methods

Seventeen cases of chronic sclerosing sialoadenitis were examined for histopathological aspects, (inflammation, fibrosis, glandular parenchyma, and lymphoid follicles) and immunohistochemistry (BCL2, CD3, CD20, CD34, CD163, p63, cyclin D1, mast cell, SMA, S100A4, IgG, and IgG4) which were scored. IgG4-related disease features were investigated. Demographic and clinical data were also collected.

Results

Males predominated (10:7), with an average lesion size of 3.9 cm. Common histopathological findings included reduced acinar parenchyma, lymphoid follicle formation, and ductular proliferation. CD3-positive T lymphocytes and CD34- and SMA-positive stromal fibroblasts were abundant. Nine cases (53%) showed sialoliths and three cases met the criteria for IgG4-related disease.

Conclusion

CSS of the submandibular gland represents a reactive pattern rather than IgG4-RD as only 3 cases seemed to be related to IgG4-RD. The immunohistochemical profile revealed an abundant population of CD3-positive T lymphocytes, as opposed to regulatory proteins such as cyclin D1, demonstrating that populations of CD34- and SMA-positive stromal fibroblasts contribute to the fibrosis characteristic of CSS. In addition, our results provide a comprehensive insight into the study of CSS and its relationship with IgG4-RD.

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Data Availability

No datasets were generated or analysed during the current study.

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Acknowledgements

The authors are grateful to CAPES and CNPq for student scholarships, and to Dr. Tayla Cruz. Albina Altemani, Ricardo Santiago Gomez, Pablo Augustin Vargas, Patrícia Ramos Cury, Felipe Paiva Fonseca, and Jean Nunes dos Santos are research fellows of the Brazilian National Council for Scientific and Technological Development (CNPq).

Funding

This work was supported by the “Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq)”; “Coordenação de Aperfeiçoamento de Ensino Superior (CAPES),” and “Fundação de Amparo à Pesquisa do Estado da Bahia (FAPESB).

Author information

Authors and Affiliations

  1. Department of Biological Sciences, State University of Feira de Santana (UEFS), Feira de Santana, Bahia, Brazil

    Vinícius Rio Verde Melo Muniz, Dandara Andrade de Santana, Larissa Abbehusen Couto, Maria Cristina Teixeira Cangussu & Patrícia Ramos Cury

  2. Department of Pathology, School of Medical Sciences, State University of Campinas, Campinas (UNICAMP), Campinas, São Paulo, Brazil

    Albina Altemani

  3. Department of Health, School of Dentistry, State University of Feira de Santana (UEFS), Feira de Santana, Feira de Santana, Bahia, Brazil

    Valéria Souza Freitas

  4. Centro de Diagnóstico Pires, Feira de Santana, Bahia, Brazil

    Bruno Cunha Pires

  5. Department of Oral Surgery and Pathology, School of Dentistry, Federal University of Minas Gerais (UFMG), Belo Horizonte, MG, Brazil

    Ricardo Santiago Gomez, Roberta Rayra Martins Chaves & Felipe Paiva Fonseca

  6. Department of Oral Pathology, University of São Paulo (USP), São Paulo, Brazil

    Suzana Catanhede Orsine Machado de Souza

  7. Oral Diagnosis Department, Piracicaba Dental School, University of Campinas (UNICAMP), Piracicaba, São Paulo, Brazil

    Pablo Augustin Vargas

  8. Department of Pathology and Forensic Medicine, School of Medicine, Federal University of Bahia (UFBA), Salvador, Bahia, Brazil

    Iguaracyra Barreto de Araújo

  9. Laboratory of Oral and Maxillofacial Pathology, School of Dentistry, Federal University of Bahia (UFBA), Salvador, Bahia, Brazil

    Jean Nunes dos Santos

  10. Faculdade de Odontologia - UFBA, Laboratório de Patologia Oral e Maxilofacial, Avenida Araújo Pinho, 62, Canela, Salvador, 40110-150, Bahia, Brazil

    Jean Nunes dos Santos

Authors
  1. Vinícius Rio Verde Melo Muniz
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Contributions

Vinícius Rio Verde Melo Muniz: Methodology; validation; investigation; formal analysis; data curation; writing (original draft); visualization; Albina Altemani: Methodology; validation; investigation; formal analysis; data curation; writing (original draft); visualization; Valéria Souza Freitas: Conceptualization; methodology; writing (review & editing); visualization; supervision. Bruno Cunha Pires: Validation; data curation; writing (review & editing). Dandara Andrade de Santana: Validation; methodology; investigation; data curation; writing (review & editing); funding acquisition. Larissa Abbehusen Couto: methodology; investigation; data curation; writing (review & editing); . Maria Cristina Teixeira Cangussu: Visualization; validation; formal analysis; writing (review & editing). Ricardo Santiago Gomez: Visualization; validation; investigation; data curation; writing (review & editing). Suzana Catanhede Orsine Machado de Souza: Visualization; validation; formal analysis; writing (review & editing). Pablo Augustin Vargas: Methodology; investigation; formal analysis; writing (review & editing); Patrícia Ramos Cury: Methodology; investigation; formal analysis; writing (review & editing); Iguaracyra Barreto de Araújo: Validation; investigation; data curation; writing (review & editing). Roberta Rayra Martins Chaves: Methodology; investigation; formal analysis; writing (review & editing); Felipe Paiva Fonseca: Visualization; validation Methodology; investigation; formal analysis; writing (review & editing); Jean Nunes dos Santos: Conceptualization; methodology; investigation; formal analysis; writing (original draft); writing (review & editing); supervision; project administration; funding acquisition.

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Correspondence to Jean Nunes dos Santos.

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All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. The study protocol was reviewed and approved by the Ethics Committee of the School of Dentistry, Federal University of Bahia (FO-UFBA) (CAAE: 45701121.6.0000.5024, Opinion number 4.834.624).

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Muniz, V.R.V.M., Altemani, A., Freitas, V.S. et al. Chronic Sclerosing Sialadenitis of the Submandibular Gland and its Histopathological Spectrum in the IgG4-Related Disease: a Series of 17 Cases. Head and Neck Pathol 18, 42 (2024). https://doi.org/10.1007/s12105-024-01651-4

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