Human Papillomavirus-Related Multiphenotypic Sinonasal Carcinoma—An Even Broader Tumor Entity?

Abstract

Human papillomavirus (HPV)-related multiphenotypic sinonasal carcinoma (HMSC) is a recently defined tumor subtype with apparent favorable clinical outcome despite aggressive histomorphology. However, in recent years, additional numbers of cases, with more variable features and at locations outside the sinonasal region, have complicated the definition of HMSC. Here, we have performed a systematic review of all cases described so far in order to accumulate more knowledge. We identified 127 articles published between 2013 and 2021, of which 21 presented unique cases. In total, 79 unique patient cases were identified and their clinical and micromorphological nature are herein summarized. In our opinion, better clinical follow-up data and a more detailed tumor characterization are preferably needed before HMSC can finally be justified as its own tumor entity.

Analysis of Human Papillomavirus (HPV) and Polyomaviruses (HPyVs) in Adenoid Cystic Carcinoma (AdCC) of the Head and Neck Region Reveals Three HPV-Positive Cases with Adenoid Cystic-like Features

“…Notably, here, we identified three patient cases, one sinonasal and two tonsillar primary tumours with a molecular and a micromorphological profile that fit within the new emerging entity of HMSC, with the exception that two of them were situated outside the sinonasal region. However, in a recent meta-analysis, including all published cases with HMSC so far, two other patients were identified as having HMSC-like tumours outside the sinonasal region [ 16 ]. Here, we added another two cases with a profile similar to HMSC, but also diagnosed outside the sinonasal region.…”

“…In 2013, however, a new additional HPV-related form of sinonasal carcinoma, now entitled HPV-related multiphenotypic sinonasal carcinoma (HMSC), was presented in a case report by Bishop and colleagues [ 15 ]. Since this first case report in 2013, fewer than 100 cases have so far been described in the literature [ 16 ]. Notably, despite the aggressive micromorphological appearance of HMSC, the diagnosis is often associated with a rather indolent clinical course.…”

“…The most common HPV type detected so far in HMSC is HPV 33, but other high-risk HPV types have also been described. Initially, HMSC was first reported to occur exclusively in the sinonasal region, but a recent case report has suggested that HMSC-like tumours may also occur outside this specific region [ 16 , 17 ].…”

Special Issue “HPV in the Head and Neck Region”

“…Recently, more efforts have also been made to disclose their effects in the head and neck region. This Special Issue on "HPV in the head and neck region" includes, one editorial, eight reviews, one brief report and two systematic reviews that contribute to the overall knowledge of HPV in the head and neck region [1][2][3][4][5][6][7][8][9][10][11][12].…”

“…A brief report, with a systematic review on HPV-related multiphenotypic sinonsasal carcinoma (HMSC), a new emerging tumor entity according to the World Health Organization (WHO) was also presented [10]. The authors indicate that this disease may possibly occur also outside the sinonasal region; however, they also suggest that HMSC needs to be better characterized before it can be justified as its own tumor entity.…”