Skip to main content

Advertisement

Log in

Signet Ring Cell Differentiation in Salivary Duct Carcinoma with Rhabdoid Features: Report of Three Cases and Literature Review

  • Case Reports
  • Published:
Head and Neck Pathology Aims and scope Submit manuscript

Abstract

Salivary duct carcinoma with rhabdoid features (SDCRF) is a rare salivary tumor with poor prognosis and is proposed as a salivary counterpart of pleomorphic lobular carcinoma of the breast (PLCB). Here, we report three cases of SDC with rhabdoid features (SDCRF) mimicking PLCB. Pleomorphic adenoma (PA) component was accompanied in all the cases confirming carcinoma ex PA. One patient had frequent rhabdoid features and showed invasive growth into the surrounding tissue. The other two patients had intracapsular tumor but with rhabdoid features. The patients with intracapsular SDCRF survived for > 5 years after surgery with no evidence of recurrence, whereas the patient with extracapsular SDCRF died 10 months after biopsy, and autopsy revealed disseminated metastasis to the central nervous system. Histologically, tumor cells in all three cases resembled PLCB, with a discohesive appearance, abundant cytoplasm, enlarged hyperchromatic nuclei, and similar immunohistochemical profiles, namely loss of membranous E-cadherin, obscured expression of membranous β-catenin, diffuse positivity of androgen receptor, gross cystic disease fluid protein-15, mitochondrial adenosine triphosphate synthase subunit β, MUC1, and INI-1. Estrogen and progesterone receptors were negative, and HER2 immunoreactivities were variable. The tumor cells of extracapsular invasive SDCRF exhibited higher MIB-1 labeling index and more frequent intracytoplasmic lumina than those of intracapsular SDCRF. Ultrastructurally, rhabdoid cells contained intracytoplasmic lumina with microvillous structure, analogous to those reported in PLCB. No intracytoplasmic intermediate filament aggregation was observed. These observations indicate that SDCRF is a salivary counterpart of PLCB and under signet ring cell differentiation.

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
Fig. 2
Fig. 3
Fig. 4
Fig. 5

Similar content being viewed by others

References

  1. Nagao T, Licitra L, Loening T, Vielh P, Williams MD. Salivary duct carcinoma. In: El-Naggar AK, Chan JKC, Rubin Grandis J, Takata T, Slootweg PJ, editors. WHO classification of head and neck tumours. Lyon: International Agency for Research on Cancer; 2017. p. 173–174.

    Google Scholar 

  2. Williams MD, Ihrler S, Seethala R. Carcinoma ex pleomorphic adenoma. In: El-Naggar AK, Chan JKC, Rubin Grandis J, Takata T, Slootweg PJ, editors. WHO classification of head and neck tumours. Lyon: International Agency for Research on Cancer; 2017. p. 176–177.

    Google Scholar 

  3. Shin S, Desmedt C, Kristiansen G, Reis-Fiho J, Sasano H. Invasive lobular carcinoma. In: Lokuhetty D, White V, Watanabe R, Cree I, editors. WHO classification of tumours breast tumours. Lyon: International Agency for Research on Cancer; 2019. p. 114–118.

    Google Scholar 

  4. Al-Baimani K, Bazzarelli A, Clemons M, Robertson SJ, Addison C, Arnaout A. Invasive pleomorphic lobular carcinoma of the breast: pathologic, clinical, and therapeutic considerations. Clin Breast Cancer. 2015;15(6):421–5. https://doi.org/10.1016/j.clbc.2015.06.010.

    Article  PubMed  Google Scholar 

  5. Chen YY, Hwang ES, Roy R, DeVries S, Anderson J, Wa C, et al. Genetic and phenotypic characteristics of pleomorphic lobular carcinoma in situ of the breast. Am J Surg Pathol. 2009;33(11):1683–94. https://doi.org/10.1097/PAS.0b013e3181b18a89.

    Article  PubMed  PubMed Central  Google Scholar 

  6. Ilic IR, Djordjevic NP, Randjelovic PJ, Stojanovic NM, Radulovic NS, Ilic RS. Seven-year survey of classical and pleomorphic invasive lobular breast carcinomas in women from southeastern Serbia: differences in clinicopathological and immunohistochemical features. J BUON. 2016;21(5):1113–20.

    PubMed  Google Scholar 

  7. Kusafuka K, Onitsuka T, Muramatsu K, Miki T, Murai C, Suda T, et al. Salivary duct carcinoma with rhabdoid features: report of 2 cases with immunohistochemical and ultrastructural analyses. Head Neck. 2014;36(3):E28–35. https://doi.org/10.1002/hed.23466.

    Article  PubMed  Google Scholar 

  8. Kusafuka K, Kawasaki T, Maeda M, Yamanegi K, Baba S, Ito Y, et al. Salivary duct carcinoma with rhabdoid features: a salivary counterpart of pleomorphic lobular carcinoma of the breast. Histopathology. 2017;70(2):164–73. https://doi.org/10.1111/his.12987.

    Article  PubMed  Google Scholar 

  9. Otsuru M, Aoki T, Kondo Y, Ota Y, Sasaki M, Suzuki T, et al. Salivary duct carcinoma with invasive micropapillary and rhabdoid feature arising in the submandibular gland. Tokai J Exp Clin Med. 2017;42(1):30–6.

    PubMed  Google Scholar 

  10. Wolff AC, Hammond ME, Hicks DG, Dowsett M, McShane LM, Allison KH, et al. Recommendations for human epidermal growth factor receptor 2 testing in breast cancer: American Society of Clinical Oncology/College of American Pathologists clinical practice guideline update. J Clin Oncol. 2013;31(31):3997–4013. https://doi.org/10.1200/JCO.2013.50.9984.

    Article  PubMed  Google Scholar 

  11. Chenevert J, Duvvuri U, Chiosea S, Dacic S, Cieply K, Kim J, et al. DOG1: a novel marker of salivary acinar and intercalated duct differentiation. Mod Pathol. 2012;25(7):919–29. https://doi.org/10.1038/modpathol.2012.57.

    Article  CAS  PubMed  Google Scholar 

  12. Loening T, Leivo I, Simpson RHW, Weinreb I. Intraductal carcinoma. In: El-Naggar AK, Chan JKC, Rubin Grandis J, Takata T, Slootweg PJ, editors. WHO classification of head and neck tumours. Lyon: International Agency for Research on Cancer; 2017. p. 170–171.

    Google Scholar 

  13. Dabbs DJ, Schnitt SJ, Geyer FC, Weigelt B, Baehner FL, Decker T, et al. Lobular neoplasia of the breast revisited with emphasis on the role of E-cadherin immunohistochemistry. Am J Surg Pathol. 2013;37(7):e1–. https://doi.org/10.1097/PAS.0b013e3182918a2b.

    Article  PubMed  Google Scholar 

  14. Okamura T, Tang XY, Saito Y, Suzuki Y, Masuda S, Tokuda Y. Pleomorphic lobular carcinoma with lipid-producing activity: a report of 2 cases. Tokai J Exp Clin Med. 2013;38(3):103–8.

    PubMed  Google Scholar 

  15. Yoshida A, Hatanaka S, Oneda S, Yoshida H. Signet ring cells in breast carcinoma. An immunohistochemical and ultrastructural study. Acta Pathol Jpn. 1992;42(7):523–8. https://doi.org/10.1111/j.1440-1827.1992.tb03100.x.

    Article  CAS  PubMed  Google Scholar 

  16. Biggs PJ, Garen PD, Powers JM, Garvin AJ. Malignant rhabdoid tumor of the central nervous system. Hum Pathol. 1987;18(4):332–7.

    Article  CAS  Google Scholar 

  17. Sobrinho-Simoes M, Johannessen JV, Gould VE. The diagnostic significance of intracytoplasmic lumina in metastatic neoplasms. Ultrastruct Pathol. 1981;2(4):327–35. https://doi.org/10.3109/01913128109081980.

    Article  CAS  PubMed  Google Scholar 

  18. Minamiguchi S, Iwasa Y, Shoji K, Higuchi K, Watanabe C, Haga H, et al. Salivary duct carcinoma: a clinicopathologic study of three cases with a review of the literature. Pathol Int. 1996;46(8):614–22. https://doi.org/10.1111/j.1440-1827.1996.tb03663.x.

    Article  CAS  PubMed  Google Scholar 

  19. Yoshihara T, Shino A, Ishii T, Kawakami M. Ultrastructural and immunohistochemical study of salivary duct carcinoma of the parotid gland. Ultrastruct Pathol. 1994;18(6):553–8. https://doi.org/10.3109/01913129409021898.

    Article  CAS  PubMed  Google Scholar 

  20. Nagao T, Sato E, Inoue R, Oshiro H, Nagai T, et al. Immunohistochemical analysis of salivary gland tumors: application for surgical pathology practice. Acta Histochem Cytochem. 2012;45(5):269–82. https://doi.org/10.1267/ahc.12019.

    Article  PubMed  PubMed Central  Google Scholar 

Download references

Acknowledgements

The authors thank Dr. Shojiroh Morinaga, Department of Diagnostic Pathology, Hino Municipal Hospital, Japan, for the diagnosis of patient 2.

Funding

No funding obtained.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Mayumi Akaki.

Ethics declarations

Conflict of interest

No conflict of interest to disclose.

Additional information

Publisher's Note

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

Electronic supplementary material

Below is the link to the electronic supplementary material.

Supplementary Table (PDF 109 kb)

Supplementary Figure (PDF 942 kb)

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Akaki, M., Ishihara, A., Nagai, K. et al. Signet Ring Cell Differentiation in Salivary Duct Carcinoma with Rhabdoid Features: Report of Three Cases and Literature Review. Head and Neck Pathol 15, 341–351 (2021). https://doi.org/10.1007/s12105-020-01186-4

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12105-020-01186-4

Keywords

Navigation