{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T12:43:17Z","timestamp":1792759397873,"version":"4.5.0"},"reference-count":40,"publisher":"MDPI AG","issue":"11","license":[{"start":{"date-parts":[[2021,5,23]],"date-time":"2021-05-23T00:00:00Z","timestamp":1621728000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000060","name":"National Institute of Allergy and Infectious Diseases","doi-asserted-by":"publisher","award":["R21AI142704"],"award-info":[{"award-number":["R21AI142704"]}],"id":[{"id":"10.13039\/100000060","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100015612","name":"CancerFree KIDS","doi-asserted-by":"publisher","award":["N\/A"],"award-info":[{"award-number":["N\/A"]}],"id":[{"id":"10.13039\/100015612","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000066","name":"National Institute of Environmental Health Sciences","doi-asserted-by":"publisher","award":["T32ES007250"],"award-info":[{"award-number":["T32ES007250"]}],"id":[{"id":"10.13039\/100000066","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Cancers"],"abstract":"<jats:p>Juvenile-onset recurrent respiratory papillomatosis (JoRRP) is the most common benign neoplasm of the larynx in children, presenting with significant variation in clinical course and potential for progression to malignancy. Since JoRRP is driven by human papillomavirus (HPV), we evaluated viral factors in a prospective cohort to identify predictive factors of disease severity. Twenty children with JoRRP undergoing routine debridement of papillomas were recruited and followed for \u22651 year. Demographical features, clinical severity scores, and surgeries over time were tabulated. Biopsies were used to establish a tissue bank and primary cell cultures for HPV6 vs. HPV11 genotyping and evaluation of viral gene expression. We found that patients with HPV11+ disease had an earlier age at disease onset, higher frequency of surgeries, increased number of lifetime surgeries, and were more likely to progress to malignancy. However, the amplitude of viral E6\/E7 gene expression did not account for increased disease severity in HPV11+ patients. Determination of HPV strain is not routinely performed in the standard of care for JoRRP patients; we demonstrate the utility and feasibility of HPV genotyping using RNA-ISH for screening of HPV11+ disease as a biomarker for disease severity and progression in JoRRP patients.<\/jats:p>","DOI":"10.3390\/cancers13112556","type":"journal-article","created":{"date-parts":[[2021,5,23]],"date-time":"2021-05-23T23:58:05Z","timestamp":1621814285000},"page":"2556","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":15,"title":["HPV Strain Predicts Severity of Juvenile-Onset Recurrent Respiratory Papillomatosis with Implications for Disease Screening"],"prefix":"10.3390","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3282-4189","authenticated-orcid":false,"given":"Mary C.","family":"Bedard","sequence":"first","affiliation":[{"name":"Division of Oncology, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alessandro","family":"de Alarcon","sequence":"additional","affiliation":[{"name":"Division of Pediatric Otolaryngology\u2013Head and Neck Surgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Otolaryngology\u2013Head and Neck Surgery, University of Cincinnati College of Medicine, Cincinnati, OH 45229, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6801-7544","authenticated-orcid":false,"given":"Yann-Fuu","family":"Kou","sequence":"additional","affiliation":[{"name":"Division of Pediatric Otolaryngology\u2013Head and Neck Surgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4040-0143","authenticated-orcid":false,"given":"David","family":"Lee","sequence":"additional","affiliation":[{"name":"Division of Pediatric Otolaryngology\u2014Head and Neck Surgery, Children\u2019s Hospital of Philadelphia, Philadelphia, PA 19104, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandra","family":"Sestito","sequence":"additional","affiliation":[{"name":"Division of Pediatric Otolaryngology\u2013Head and Neck Surgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Angela L.","family":"Duggins","sequence":"additional","affiliation":[{"name":"Division of Pediatric Otolaryngology\u2013Head and Neck Surgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marion","family":"Brusadelli","sequence":"additional","affiliation":[{"name":"Division of Clinical Operations, Medpace, Cincinnati, OH 45227, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adam","family":"Lane","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA"},{"name":"Division of Bone Marrow Transplantation and Immune Deficiency, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathryn A.","family":"Wikenheiser-Brokamp","sequence":"additional","affiliation":[{"name":"Division of Pathology &amp; Laboratory Medicine and The Perinatal Institute Division of Pulmonary Biology, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Pathology &amp; Laboratory Medicine, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Susanne I.","family":"Wells","sequence":"additional","affiliation":[{"name":"Division of Oncology, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH 45267, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0048-4012","authenticated-orcid":false,"given":"David F.","family":"Smith","sequence":"additional","affiliation":[{"name":"Division of Pediatric Otolaryngology\u2013Head and Neck Surgery, Cincinnati Children\u2019s Hospital Medical Center, Cincinnati, OH 45229, USA"},{"name":"Department of Otolaryngology\u2013Head and Neck Surgery, University of Cincinnati College of Medicine, Cincinnati, OH 45229, USA"},{"name":"Division of Pulmonary and Sleep Medicine, Cincinnati Children\u2019s Hospital Medical Center, 3333 Burnet Ave. MLC 2018, Cincinnati, OH 45229, USA"},{"name":"Center for Circadian Medicine, Cincinnati Children\u2019s Hospital Medical Center, 3333 Burnet Ave. MLC 2018, Cincinnati, OH 45229, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2021,5,23]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"1236","DOI":"10.1097\/MLG.0b013e31816a7135","article-title":"Recurrent Respiratory Papillomatosis: A Review","volume":"118","author":"Derkay","year":"2008","journal-title":"Laryngoscope"},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1386","DOI":"10.1001\/archotol.1995.01890120044008","article-title":"Task Force on Recurrent Respiratory Papillomas: A Preliminary Report","volume":"121","author":"Derkay","year":"1995","journal-title":"Arch. 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