An Uncommon Cause of Dyspnea: Tracheal Papillomatosis

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Abstract Introduction: Tracheal papillomatosis (TP) is a rare disease with predilection for the upper airway in response to human papilloma virus (HPV) infection. It exhibits a bimodal age distribution with the adult form being less prevalent. The most common sites where lesions are identified is the larynx, and less commonly on the trachea. The risk of malignant transformation to squamous cell carcinoma (SCC) is rare and usually seen in adults with an incidence of around 3-7%. Here we present a patient with dyspnea and shortness of breath (SOB) secondary to TP with progression to SCC. Case presentation: A 77-year-old male former smoker of 90 pack years was consulted to the pulmonary service due to dyspnea and SOB. Chest CT was found with multiple endotracheal nodularity's originating from the inner tracheal wall protruding into the lumen raising suspicion for a malignant process. Patient denied chills, cough, fever, sputum production or weight loss. Flexible bronchoscopy was performed and showed multiple friable exophytic tan-pink cauliflower like lesions mostly on the anterior and posterior aspect of the trachea. Biopsies were remarkable for squamous papilloma with high grade dysplasia suggestive of diffuse TP. HPV low/high risk RNA in situ hybridization was negative for HPV sub-types 6, 11, 16, 18, 31, 33 and 51. Tracheal lesions were resected through electrical snare followed by argon plasma coagulation ablation with resected tissue consisting of papillary structures with hyperplastic squamous epithelium with fibrovascular cores and scattered koilocytic atypia. These morphologic features were consistent with squamous papilloma. Discussion: TP lesions tend to be slow-growing and have a high recurrence rate after treatment. HPV types 6 and 11 are the most common identified subtypes followed by subtypes 16 and 18 which are associated with poorer outcomes. Clinical presentation is non-specific however dissemination and progression of disease may lead to cough, dyspnea, SOB, and upper airway obstruction with respiratory failure. Degeneration to SCC leads to poorer prognosis. It is usually associated to high-risk HPV genotypes and smoking. Due to the rarity of the condition, there is no therapeutic guidelines however in the presence of malignant degeneration excision of the papilloma is fundamental. Widespread HPV vaccination is expected to decrease future incidence of respiratory papillomatosis with the goal of eradicating this disease, nevertheless it remains a public health issue. With this case we aim to highlight the importance of identifying respiratory papilloma to prevent symptoms and conversion to SCC.

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