{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,28]],"date-time":"2026-03-28T00:49:21Z","timestamp":1774658961383,"version":"3.50.1"},"reference-count":13,"publisher":"Georg Thieme Verlag KG","issue":"01","license":[{"start":{"date-parts":[[2026,3,27]],"date-time":"2026-03-27T00:00:00Z","timestamp":1774569600000},"content-version":"vor","delay-in-days":85,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Neurol Surg Rep"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Human papillomavirus (HPV)\u2013related multiphenotypic sinonasal carcinoma (HMSC) is a rare neoplasm characterized by a paradoxical clinical course. Despite its high-grade histologic features, HMSC exhibits an unexpectedly indolent clinical behavior, marked by frequent local recurrences but limited metastatic potential. We report the first case of HMSC metastasis to the liver with the earliest known distant progression.<\/jats:p>\n                  <jats:p>A 53-year-old male who presented with unilateral epistaxis was found to have T4aN0M0 HPV35-positive HMSC of the left maxillary sinus, with erosion into the orbital floor. He underwent induction chemotherapy, endoscopic resection with negative margins, and adjuvant chemoradiation. He did well until surveillance PET-CT at 10 months revealed asymptomatic hepatic metastases confirmed by biopsy. He was treated with transarterial chemoembolization and pembrolizumab before developing locoregional recurrence involving the orbital apex and cavernous sinus 5 months later.<\/jats:p>\n                  <jats:p>Several case reports demonstrate HMSC's potential for aggressive behavior, including intracranial extension and rapid recurrence. However, distant metastases (DM) are rare, with only three documented cases of pulmonary metastases and one digital metastasis, occurring years after initial treatment. We present the first reported case of HMSC with metastasis to the liver, marking the earliest known distant recurrence. This case challenges the current paradigm by highlighting HMSC's potential for aggressive systemic progression. Molecularly, this tumor harbored high-risk HPV35, a rarely reported subtype. Despite HMSC's typically indolent course, this case underscores the importance of vigilant surveillance to detect early DM and guide therapeutic management.<\/jats:p>","DOI":"10.1055\/a-2773-6796","type":"journal-article","created":{"date-parts":[[2026,3,27]],"date-time":"2026-03-27T23:53:32Z","timestamp":1774655612000},"page":"e59-e61","source":"Crossref","is-referenced-by-count":0,"title":["Metastatic HPV-Related Multiphenotypic Sinonasal Carcinoma: A Case Report and Review of the Literature"],"prefix":"10.1055","volume":"87","author":[{"given":"Rachel","family":"Stemme","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology-Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wassim","family":"Najjar","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology-Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tanguy Y.","family":"Seiwert","sequence":"additional","affiliation":[{"name":"Department of Oncology, Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Harry","family":"Quon","sequence":"additional","affiliation":[{"name":"Department of Radiation Oncology and Molecular Radiation Sciences, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lisa M.","family":"Rooper","sequence":"additional","affiliation":[{"name":"Department of Oncology, Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"},{"name":"Department of Pathology, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Debraj","family":"Mukherjee","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1296-2648","authenticated-orcid":false,"given":"Nicholas R.","family":"Rowan","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology-Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"},{"name":"Department of Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"194","published-online":{"date-parts":[[2026,3,27]]},"reference":[{"issue":"02","key":"ref1","doi-asserted-by":"crossref","first-page":"185","DOI":"10.1097\/PAS.0b013e3182698673","article-title":"Human papillomavirus-related carcinomas of the sinonasal tract","volume":"37","author":"J A Bishop","year":"2013","journal-title":"Am J Surg Pathol"},{"issue":"12","key":"ref2","doi-asserted-by":"crossref","first-page":"1690","DOI":"10.1097\/PAS.0000000000000944","article-title":"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","volume":"41","author":"J A Bishop","year":"2017","journal-title":"Am J Surg Pathol"},{"issue":"09","key":"ref3","doi-asserted-by":"crossref","first-page":"1861","DOI":"10.3390\/v13091861","article-title":"Human papillomavirus-related multiphenotypic sinonasal carcinoma-an even broader tumor entity?","volume":"13","author":"M Zupancic","year":"2021","journal-title":"Viruses"},{"issue":"04","key":"ref4","doi-asserted-by":"crossref","first-page":"e41","DOI":"10.1055\/s-0039-3399571","article-title":"Human papillomavirus related multiphenotypic sinonasal carcinoma: report of a case with early and progressive metastatic disease","volume":"80","author":"A I Rodarte","year":"2019","journal-title":"J Neurol Surg Rep"},{"issue":"04","key":"ref5","doi-asserted-by":"crossref","first-page":"599","DOI":"10.1016\/j.path.2024.07.002","article-title":"Human papillomavirus in sinonasal tract tumors-another head and neck \u201chot spot\u201d for prognostically favorable viral disease","volume":"17","author":"J S Lewis Jr","year":"2024","journal-title":"Surg Pathol Clin"},{"issue":"07","key":"ref6","doi-asserted-by":"crossref","first-page":"609","DOI":"10.1001\/jamaoto.2024.1311","article-title":"Epidemiologic trends in human papillomavirus-associated 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Kuroda","year":"2022","journal-title":"Neuropathology"},{"issue":"09","key":"ref10","first-page":"e68824","article-title":"Human papillomavirus (HPV)-related multiphenotypic sinonasal carcinoma with intracranial extension and rapid recurrence: a case report","volume":"16","author":"A N Reinders","year":"2024","journal-title":"Cureus"},{"issue":"02","key":"ref11","doi-asserted-by":"crossref","first-page":"258","DOI":"10.1158\/1078-0432.CCR-24-1416","article-title":"Phase II trial of induction chemotherapy for advanced sinonasal squamous cell carcinoma","volume":"31","author":"K J Contrera","year":"2025","journal-title":"Clin Cancer Res"},{"issue":"07","key":"ref12","doi-asserted-by":"crossref","first-page":"1226","DOI":"10.1002\/alr.23380","article-title":"The role of induction chemotherapy for orbital invasion in sinonasal malignancies: a systematic review","volume":"14","author":"A Tang","year":"2024","journal-title":"Int Forum Allergy Rhinol"},{"key":"ref13","doi-asserted-by":"crossref","first-page":"106555","DOI":"10.1016\/j.oraloncology.2023.106555","article-title":"Induction chemotherapy response of HPV-related multiphenotypic sinonasal carcinoma: first-ever reported therapeutic outcome and a brief literature review","volume":"146","author":"G S Vieira","year":"2023","journal-title":"Oral Oncol"}],"container-title":["Journal of Neurological Surgery 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