Head and Neck Pathology

, Volume 12, Issue 4, pp 623–628 | Cite as

Human Papillomavirus-Related Multiphenotypic Sinonasal Carcinoma: A Case Report Documenting the Potential for Very Late Tumor Recurrence

  • Akeesha A. ShahEmail author
  • Eric D. Lamarre
  • Justin A. Bishop
Case Report


Human papillomavirus (HPV)-related multiphenotypic sinonasal carcinoma is a peculiar sinonasal tract tumor that demonstrates features of both a surface-derived and salivary gland carcinoma. Implicit in its name, this tumor has a consistent association with high-risk HPV, particularly type 33. It was first described in 2013 under the designation of HPV-related carcinoma with adenoid cystic carcinoma-like features. However, since its initial description additional cases have emerged which demonstrate a wide morphologic spectrum and relatively indolent clinical behavior. Herein we report our experience with a case of HPV-related multiphenotypic sinonasal carcinoma that was initially classified as adenoid cystic carcinoma in the 1980s. The patient recurred after a 30-year disease free interval. RNA in situ hybridization confirmed the presence of high-risk HPV in both her recurrence and her initial tumor in the 1980s, which allowed for reclassification as HPV-related multiphenotypic sinonasal carcinoma. Our case adds to the literature of this relatively newly described entity and supports the indolent clinical behavior of this neoplasm but also demonstrates a potential for very late local recurrence.


Human papillomavirus Multiphenotypic sinonasal carcinoma Adenoid cystic carcinoma Sinonasal carcinoma Carcinoma with adenoid cystic-like features 


  1. 1.
    Bishop JA, Guo TW, Smith DF, et al. Human papillomavirus-related carcinomas of the sinonasal tract. Am J Surg Pathol. 2013;37:185–92.CrossRefGoogle Scholar
  2. 2.
    Bishop JA, Ogawa T, Stelow EB, 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.CrossRefGoogle Scholar
  3. 3.
    Andreasen S, Bishop JA, Hansen TV, et al. Human papillomavirus-related carcinoma with adenoid cystic-like features of the sinonasal tract: clinical and morphological characterization of six new cases. Histopathology. 2017;70:880–8.CrossRefGoogle Scholar
  4. 4.
    Hwang SJ, Ok S, Lee HM, et al. Human papillomavirus-related carcinoma with adenoid cystic-like features of the inferior turbinate: a case report. Auris Nasus Larynx. 2015;42:53–5.CrossRefGoogle Scholar
  5. 5.
    Bishop JA, Andreasen S, Hang JF, 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.CrossRefGoogle Scholar
  6. 6.
    El-Naggar AK, Chen JCK, Grandis JR, Takata T, Slootweg PJ, editors. World Health Organization classification of head and neck tumours. 4th ed. Lyon: IARC Press; 2017.Google Scholar
  7. 7.
    Bishop JA, Ma XJ, Wang H, et al. 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;36:1874–82.CrossRefGoogle Scholar
  8. 8.
    Seethala RR, Hunt JL, Baloch ZW, et al. Adenoid cystic carcinoma with high-grade transformation: a report of 11 cases and a review of the literature. Am J Surg Pathol. 2007;31:1683–94.CrossRefGoogle Scholar

Copyright information

© Springer Science+Business Media, LLC, part of Springer Nature 2018

Authors and Affiliations

  • Akeesha A. Shah
    • 1
    Email author
  • Eric D. Lamarre
    • 2
  • Justin A. Bishop
    • 3
  1. 1.Department of Pathology, Cleveland Clinic FoundationRobert J. Tomsich Pathology and Laboratory Medicine InstituteClevelandUSA
  2. 2.Head and Neck InstituteCleveland Clinic FoundationClevelandUSA
  3. 3.Department of PathologyUniversity of Texas Southwestern Medical CenterDallasUSA

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