Abstract
BackgroundRecurrent respiratory papillomatosis is a chronic disease of viral origin affecting the larynx, trachea, and lower airways. Inverted papilloma, most commonly originating from the lateral nasal wall, is typically a single, expansile, locally aggressive tumor that remodels bone around the site of origin.Case presentationWe report a case of histopathologically proven inverted papilloma occurring in a 50-year-old Caucasian man with recurrent respiratory papillomatosis affecting his nasal cavity, larynx, and trachea. This constitutes the first report of nasal involvement in recurrent respiratory papillomatosis. Viral in situ hybridization studies demonstrated evidence of human papillomavirus in both the septum and middle turbinate subsites. Repeat nasal excision with margin analysis is planned.ConclusionsThis report emphasizes the importance of considering a broad differential diagnosis in patients with papillomata, and obtaining comprehensive histopathologic evaluation of lesions in multiple subsites in order to rule out inverted papilloma or overt malignant transformation, particularly if high-risk human papillomavirus (HPV) subtypes are identified.Level of evidence4
Highlights
Recurrent respiratory papillomatosis (RRP) is a chronic disease of viral origin most commonly affecting squamous and ciliated epithelia of the larynx, trachea, and lower airways, at the laryngeal ventricle and carina [1]
Common symptoms of RRP range from voice changes to life-threatening airway compromise, Oliver et al Journal of Medical Case Reports (2019) 13:215 often requiring repeat endoscopic procedures to remove the obstructing lesions and preserve normal tissue and function [3]. We present this case of histopathologically proven inverted papilloma (IP) occurring in a patient with RRP affecting the nasal cavity, larynx, and trachea
The rate of dysplasia in patients with adult-onset RRP is roughly 10%, with malignancy developing in up to 5%; rates of dysplasia can reach as high as 55% in recent studies reported in the literature; some authors suggest that human papillomavirus (HPV)-negative papilloma, tobacco use, and previous cidofovir administration may increase the risk of malignant transformation [4]
Summary
This represents the first report of IP occurring within the context of RRP. Interestingly, the same strain of HPV was isolated in both the IP and RRP lesions. Our case provides support to the theory of HPV involvement in the pathogenesis of both IP and RRP lesions. We were unable to find any additional reported cases of diffuse sinonasal papillomata. Carcinoma ex-Schneiderian papilloma (malignant transformation): a clinicopathologic and immunophenotypic study of 20 cases combined with a comprehensive review of the literature. JS and DE participated in the design of the study and edited the manuscript text and image panels. Ethics approval and consent to participate Ethics approval and consent to publish was obtained from the patient in this case report through our institutional consent forms. This study was deemed Institutional Review Board (IRB) exempt by the Mayo Clinic IRB. Consent for publication Written informed consent was obtained from the patient for publication of this case report and any accompanying images. Author details 1Mayo Clinic School of Medicine, Mayo Clinic, Rochester, MN, USA. 2Department of Otorhinolaryngology, Mayo Clinic, 200 1st St SW, Rochester, MN 55905, USA
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