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

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.

Human Papillomavirus Related Multiphenotypic Sinonasal Carcinoma: Report of a Case with Early and Progressive Metastatic Disease

“…In the limited cases that recur, the tumors are slow growing, with the timing of recurrence ranging widely: the earliest local recurrence previously reported occurred 23 months posttreatment, and the latest recurrence previously reported occurred after a 30-year disease-free interval. 1 3 4 6 7 8 9 10 11 Most previously reported recurrences responded well to treatment, achieving long disease-free intervals. Only two cases previously reported progressed to distal metastasis after 96 and 144 months following their initial treatment.…”

“…3 6 Most HPV-positive head and neck carcinomas arise from the oropharynx, with the sinonasal tract now recognized as another anatomic “hotspot” for HPV-related neoplasms. 3 10 14 Sinonasal tract HMSC is most often associated with HPV type 33. 4 Here, we describe the case of an HMSC with HPV-16 identified by both RT-PCR (reverse transcription polymerase chain reaction) and HPV-16/18 RNA in situ hybridization associated with an aggressive clinical course.…”

“…Most reports published since this series have followed this trend. 1 3 4 6 7 8 9 10 To our knowledge, only a few recorded cases have documented locally aggressive tumors or very late distant metastasis, and there have been no reported tumor-related deaths. 3 11 12 Here, we present a case of HMSC with early and progressive metastatic disease.…”

Human Papillomavirus (HPV) Infection in Males: A Need for More Awareness

“…Although there were studies suggesting possible association between HPV and prostate cancer in males, none have reached universal acceptability [52,53]. Recently, a causal association between hrHPV and HPV-related multiphenotypic sinonasal carcinoma (HMSC) has been described [54][55][56][57]. Other malignancies associated with HPV include SCC of the skin/nose tip and skin appendages [58][59][60][61][62].…”

Morphological Changes of the Endometrium in Hyperplastic Process in Women of Reproductive Age

“…A number of authors consider HPE as a clinical and morphological syndrome caused by damage to the endometrium by pathogen infections, with numerous secondary morphological and functional changes that disrupt cyclic biotransformation and susceptibility of the mucous membrane of the uterus [10][11][12][13][14]. Other scientists also note the role of the infectious factor in the occurrence of EH despite the lack of growth of the microflora in crops from the uterine cavity [15][16][17][18][19].…”