Skip to main content
SpringerLink
Log in

Human Papillomavirus-Related Multiphenotypic Sinonasal Carcinoma with Unique HPV type 52 Association: A Case Report with Review of Literature

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

Abstract

Human papillomavirus (HPV)-related multiphenotypic sinonasal carcinoma (HMSC) is a recently described distinctive clinicopathologic entity defined by association to high risk HPV, localization to sinonasal tract and close histologic resemblance to salivary gland tumors. Lack of awareness of its pathologic features and biology among pathologists and oncologists make this entity susceptible to misdiagnosis and erroneous management. Herein, we illustrate a case of HMSC of the nasal cavity associated with heretofore unreported subtype HPV-52 and discuss the challenges associated with diagnosis and management of this rare tumor. A 48-year-old woman with intermittent epistaxis for 6 months presented with a nasal mass and underwent middle turbinectomy. Histology showed a tumor with features typical of adenoid cystic carcinoma (ACC) in the form of basaloid cells and cribriform architecture. However, careful inspection revealed findings uncommon in ACC; such as surface pagetoid tumor spread, areas of solid sheets of myoepithelial cells accompanied by increased mitotic figures which prompted immunohistochemistry. Multidirectional differentiation into ductal (CK7, AE1/AE3) and myoepithelial (p63, p40, S100, calponin) lineage together with strong and diffuse immunopositivity for p16 distinguished this tumor from ACC. HPV genotyping was positive for high risk HPV subtype HPV52, which confirmed the diagnosis of HMSC. HPV-related multiphenotypic sinonasal carcinoma is an under-recognized unique clinicopathologic entity that needs awareness to avoid mistaking it for commoner salivary gland tumors. Making accurate diagnosis of this newly-described tumor is imperative in order to understand its biology and to develop optimal therapeutic strategies.

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

Access this article

Log in via an institution

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

Similar content being viewed by others

References

  1. Singhi AD, Westra WH. Comparison of human papillomavirus in situ hybridization and p16 immunohistochemistry in the detection of human papillomavirus-associated head and neck cancer based on a prospective clinical experience. Cancer. 2010;116:2166–73.

    PubMed  Google Scholar 

  2. Stevens TM, Bishop JA. HPV-related carcinomas of the head and neck: morphologic features, variants, and practical considerations for the surgical pathologist. Virchows Arch. 2017;471(2):295–307.

    Article  PubMed  Google Scholar 

  3. Bishop JA, Andreasen S, Hang JF, Bullock MJ, Chen TY, Franchi A, et al. HPV-related multiphenotypic sinonasal carcinoma: an expanded series of 49 cases of the tumor formerly known as HPV-related carcinoma with adenoid cystic carcinoma-like features. Am J Surg Pathol. 2017;41:1690–701.

    Article  PubMed  PubMed Central  Google Scholar 

  4. Shah AA, Lamarre ED, Bishop JA. Human papillomavirus-related multiphenotypic sinonasal carcinoma: a case report documenting the potential for very late tumor recurrence. Head Neck Pathol. 2018. https://doi.org/10.1007/s12105-018-0895-5.

    Article  PubMed  PubMed Central  Google Scholar 

  5. Ruangritchankul K, Jitpasutham T, Kitkumthorn N, Thorner PS, Keelawat S. Human papillomavirus-related multiphenotypic sinonasal carcinoma: first case report associated with an intermediate-risk HPV type and literatures review. Hum Pathol. 2018;14:20–4.

    Google Scholar 

  6. Hwang SJ, Ok S, Lee HM, Lee E, Park IH. Human papillomavirus-related carcinoma with adenoid cystic-like features of the inferior turbinate: a case report. Auris Nasus Larynx. 2015;42:53–5.

    Article  PubMed  Google Scholar 

  7. Andreasen S, Bishop JA, Hansen TV, Westra WH, Bilde A, von Buchwald C, et al. Human papillomavirus-related carcinoma with adenoid cystic-like features of the sinonasal tract: clinical and morphological characterization of six new cases. Histopathol. 2017;70:880–8.

    Article  Google Scholar 

  8. Bishop JA, Ogawa T, Stelow EB, Moskaluk CA, Koch WM, Pai SI, et al. Human papillomavirus-related carcinoma with adenoid cystic-like features: a peculiar variant of head and neck cancer restricted to the sinonasal tract. Am J Surg Pathol. 2013;37:836–44.

    Article  PubMed  PubMed Central  Google Scholar 

  9. Hang JF, Hsieh MS, Li WY, Chen JY, Lin SY, Liu SH, et al. Human papillomavirus-related carcinoma with adenoid cystic-like features: a series of five cases expanding the pathological spectrum. Histopathology. 2017;71(6):887–896.

    Article  PubMed  Google Scholar 

  10. Lewis JS, Beadle B, Bishop JA, Chernock RD, Colasacco C, Lacchetti C, et al. Human papillomavirus testing in head and neck carcinomas: guideline from the college of american pathologists. Arch Pathol Lab Med. 2018;142(5):559–97.

    Article  PubMed  Google Scholar 

  11. Boland JM, McPhail ED, Garcia JJ, et al. Detection of human papilloma virus and p16 expression in high-grade adenoid cystic carcinoma of the head and neck. Mod Pathol. 2012;25:529–36.

    Article  PubMed  Google Scholar 

  12. Jour G, West K, Ghali V, Shank D, Ephrem G, Wenig BM. Differential expression of p16(INK4A) and cyclin D1 in benign and malignant salivary gland tumors: a study of 44 Cases. Head Neck Pathol. 2013;7(3):224–31.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  13. Bishop JA, Ma XJ, Wang H, Luo Y, Illei PB, Begum S, Taube JM, Koch WM, Westra WH. Detection of transcriptionally active high-risk HPV in patients with head and neck squamous cell carcinoma as visualized by a novel E6/E7 mRNA in situ hybridization method. Am J Surg Pathol. 2012;36(12):1874–82.

    Article  PubMed  PubMed Central  Google Scholar 

  14. Isayeva. T, Li Y, Maswahu, D, Brandwein-Gensler M. Human papillomavirus in non-oropharyngeal head and neck cancers: a systematic literature review. Head Neck Pathol. 2012;6:104–20.

    Article  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Pathology, Tata Memorial Hospital, Parel, Mumbai, Maharashtra, 400012, India

    Shraddha A. Adamane, Neha Mittal & Munita Bal

  2. Teni Lab, Advanced Centre for Treatment, Research and Education in Cancer (ACTREC), Kharghar, Navi Mumbai, 410 210, India

    Tanuja Teni, Sagar Pawar & Rohit Waghole

Authors
  1. Shraddha A. Adamane
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Neha Mittal
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Tanuja Teni
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Sagar Pawar
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Rohit Waghole
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Munita Bal
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Munita Bal.

Ethics declarations

Conflict of interest

All authors declare no conflicts of interest.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Adamane, S.A., Mittal, N., Teni, T. et al. Human Papillomavirus-Related Multiphenotypic Sinonasal Carcinoma with Unique HPV type 52 Association: A Case Report with Review of Literature. Head and Neck Pathol 13, 331–338 (2019). https://doi.org/10.1007/s12105-018-0969-4

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12105-018-0969-4

Keywords

Search

Navigation