Skip to main content

Advertisement

Log in

Basal Cell Adenoma and Basal Cell Adenocarcinoma: a 50-year Experience From a Single Institution

  • Original Paper
  • Published:
Head and Neck Pathology Aims and scope Submit manuscript

Abstract

Background

Basal cell adenoma (BCA) and adenocarcinoma (BCAd) are two of the least frequent salivary gland tumors. We describe the largest series of these neoplasms, spanning over a period of 50 years (1970–2020), diagnosed and treated in a single Institution.

Methods

Sixty-eight cases were identified. Clinical and pathological data were collected and correlated with outcome.

Results

Forty-one BCA and 27 BCAd were identified. BCA cases had almost pristine prognosis, with only a relapse in a tumor inadequately excised. Ten patients with BCAd developed metastases, and 14 died from the disease. The 2-year and 5-year survival was of 76% and 42%.

Conclusions

The importance of adequate excision is reinforced in BCA, with no recurrences occurring when margins were negative. Contrary to previous reports, BCAd was not associated with a good prognosis. A better understanding of the genetics of these neoplasms may identify therapeutic options when dealing with inoperable or metastatic disease.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Graph 1
Graph 2
Graph 3

Similar content being viewed by others

Data Availability

Not applicable.

Code Availability

Not applicable.

References

  1. WHO Classification of Head and Neck Tumours [Internet; beta version ahead of print] [Internet]. (2022) 5th ed. Lyon (France): International Agency for Research on Cancer. https://tumourclassification.iarc.who.int/chapters/52. Accessed 10 July 2022

  2. Muller S, Barnes L. Basal cell adenocarcinoma of the salivary glands: report of seven cases and review of the literature. Cancer Interdiscip Int J Am Cancer Soc. 1996;78:2471–7.

    CAS  Google Scholar 

  3. Zhan KY, Lentsch EJ. Basal cell adenocarcinoma of the major salivary glands: a population-level study of 509 cases. Laryngoscope. 2016;126:1086–90.

    Article  Google Scholar 

  4. Luna MA, Batsakis JG, El-Naggar AK. Basaloid monomorphic adenomas. Ann Otol Rhinol Laryngol. 1991;100:687–90.

    Article  Google Scholar 

  5. Chen KT. Carcinoma arising in monomorphic adenoma of the salivary gland. Am J Otolaryngol. 1985;6:39–41.

    Article  CAS  Google Scholar 

  6. Luna MA, Batsakis JG, Tortoledo ME, Del Junco GW. Carcinomas ex monomorphic adenoma of salivary glands. J Laryngol Otol. 1989;103:756–9.

    Article  CAS  Google Scholar 

  7. Rito M, Fonseca I. Carcinoma ex-pleomorphic adenoma of the salivary glands has a high risk of progression when the tumor invades more than 2.5 mm beyond the capsule of the residual pleomorphic adenoma. Virchows Arch. 2016. https://doi.org/10.1007/s00428-015-1887-4.

    Article  Google Scholar 

  8. Rito M, Mitani Y, Bell D, Mariano FV, Almalki ST, Pytynia KB, et al. Frequent and differential mutations of the CYLD gene in basal cell salivary neoplasms: linkage to tumor development and progression. Mod Pathol. 2018. https://doi.org/10.1038/s41379-018-0018-6.

    Article  Google Scholar 

  9. Wilson TC, Ma D, Tilak A, Tesdahl B, Robinson RA. Next-generation sequencing in salivary gland basal cell adenocarcinoma and basal cell adenoma. Head Neck Pathol. 2016;10:494–500.

    Article  Google Scholar 

  10. Kazakov DV. Brooke-Spiegler syndrome and phenotypic variants: an update. Head Neck Pathol. 2016;10:125–30.

    Article  Google Scholar 

  11. Fonseca I, Soares J. Basal cell adenocarcinoma of minor salivary and seromucous glands of the head and neck region. Semin Diagn Pathol. 1996;13(2):128–37.

    CAS  Google Scholar 

  12. Amin MB, Edge S, Greene F, Byrd DR, Brookland RK, Washington MK, Gershenwald JE, Compton CC, Hess KR, et al. (Eds.). AJCC Cancer Staging Manual (8th edition). Springer International Publishing: American Joint Commission on Cancer. 2017.

  13. Junquera L, Gallego L, de Vicente JC, Fresno MF. Bilateral parotid basal cell adenoma: an unusual case report and review of the literature. J Oral Maxillofac Surg. 2010;68:179–82.

    Article  Google Scholar 

  14. Ethunandan M, Pratt CA, Morrison A, Anand R, Macpherson DW, Wilson AW. Multiple synchronous and metachronous neoplasms of the parotid gland: the Chichester experience. Br J Oral Maxillofac Surg. 2006;44:397–401.

    Article  CAS  Google Scholar 

  15. Myssiorek D, Ruah CB, Hybels RL. Recurrent pleomorphic adenomas of the parotid gland. Head Neck. 1990;12:332–6.

    Article  CAS  Google Scholar 

  16. Robinson RA. Basal cell adenoma and basal cell adenocarcinoma. Surg Pathol Clin. 2021;14:25–42.

    Article  Google Scholar 

  17. Shingaki S, Ohtake K, Nomura T, Nakajima T. The role of radiotherapy in the management of salivary gland carcinomas. J Cranio-Maxillofacial Surg. 1992;20:220–4.

    Article  CAS  Google Scholar 

  18. Nagao T, Sugano I, Ishida Y, Hasegawa M, Matsuzaki O, Konno A, et al. Basal cell adenocarcinoma of the salivary glands: comparison with basal cell adenoma through assessment of cell proliferation, apoptosis, and expression of p53 and bcl-2. Cancer Interdiscip Int J Am Cancer Soc. 1998;82:439–47.

    CAS  Google Scholar 

  19. Schmitt AC, Griffith CC, Cohen C, Siddiqui MT. LEF-1: diagnostic utility in distinguishing basaloid neoplasms of the salivary gland. Diagn Cytopathol. 2017;45:1078–83.

    Article  Google Scholar 

  20. Bilodeau EA, Acquafondata M, Barnes EL, Seethala RR. A comparative analysis of LEF-1 in odontogenic and salivary tumors. Hum Pathol. 2015;46:255–9.

    Article  CAS  Google Scholar 

  21. Kawahara A, Harada H, Abe H, Yamaguchi T, Taira T, Nakashima K, et al. Nuclear β-catenin expression in basal cell adenomas of salivary gland. J Oral Pathol Med. 2011;40:460–6.

    Article  CAS  Google Scholar 

  22. Jo VY, Sholl LM, Krane JF. Distinctive patterns of CTNNB1 (β-catenin) alterations in salivary gland basal cell adenoma and basal cell adenocarcinoma. Am J Surg Pathol. 2016;40:1143–50.

    Article  Google Scholar 

  23. Lee Y-H, Huang W-C, Hsieh M-S. CTNNB1 mutations in basal cell adenoma of the salivary gland. J Formos Med Assoc. 2018;117:894–901.

    Article  CAS  Google Scholar 

  24. Sato M, Yamamoto H, Hatanaka Y, Nishijima T, Jiromaru R, Yasumatsu R, et al. Wnt/β-catenin signal alteration and its diagnostic utility in basal cell adenoma and histologically similar tumors of the salivary gland. Pathol Pract. 2018;214:586–92.

    Article  CAS  Google Scholar 

  25. Jung MJ, Roh J-L, Choi S-H, Nam SY, Kim SY, Lee S, et al. Basal cell adenocarcinoma of the salivary gland: a morphological and immunohistochemical comparison with basal cell adenoma with and without capsular invasion. Diagn Pathol. 2013;8:1–8.

    Article  Google Scholar 

  26. Geiger JL, Ismaila N, Beadle B, Caudell JJ, Chau N, Deschler D, et al. Management of salivary gland malignancy: ASCO guideline. J Clin Oncol. 2021. https://doi.org/10.1200/JCO.21.00449.

    Article  Google Scholar 

  27. National Comprehensive Cancer Network (2021) NCCN Guidelines Head and Neck [Internet]. https://www.nccn.org/professionals/physician_gls/pdf/head-and-neck.pdf. Accessed 10 July 2022

  28. Gustin JP, Cosgrove DP, Park BH. The PIK3CA gene as a mutated target for cancer therapy. Curr Cancer Drug Targets. 2008;8:733–40.

    Article  CAS  Google Scholar 

  29. FDA (2019) FDA approves alpelisib for metastatic breast cancer [Internet]. https://www.fda.gov/drugs/resources-information-approved-drugs/fda-approves-alpelisib-metastatic-breast-cancer. Accessed 10 July 2022

  30. Rajan N, Elliott R, Clewes O, Mackay A, Reis-Filho JS, Burn J, et al. Dysregulated TRK signalling is a therapeutic target in CYLD defective tumours. Oncogene Nature Publishing Group. 2011;30:4243–60.

    CAS  Google Scholar 

Download references

Funding

No funding was received for conducting this study.

Author information

Authors and Affiliations

Authors

Contributions

MR and IF: Contributed to the study conception and design, reviewed the pathological slides and agreed on the diagnosis. MR: performed the data collection, data analysis and wrote the first draft of the manuscript. Susana Esteves did the statistical analysis. Isabel Fonseca and Susana Esteves reviewed and edited the manuscript. All authors read and approved the final manuscript.

Corresponding author

Correspondence to Miguel Rito.

Ethics declarations

Conflict of interest

The authors have no relevant financial or non-financial interests to disclose.

Ethical Approval

The study was approved by the Institutional board of Instituto Português de Oncologia de Lisboa Francisco Gentil (Lisboa, Portugal), project number UIC/993.

Research Involving Human and Animals Rights

Not applicable.

Consent to participate

Not applicable.

Consent for publication

Not applicable.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Springer Nature or its licensor holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Rito, M., Esteves, S. & Fonseca, I. Basal Cell Adenoma and Basal Cell Adenocarcinoma: a 50-year Experience From a Single Institution. Head and Neck Pathol 16, 1157–1166 (2022). https://doi.org/10.1007/s12105-022-01484-z

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12105-022-01484-z

Keywords

Navigation