Abstract Introduction Recurrent respiratory papillomatosis (RRP) is a rare but relentlessly recurrent airway disease caused by human papillomavirus (HPV), most commonly types 6 and 11. Adult-onset RRP typically follows an indolent yet persistent course, with repeated recurrences leading to airway obstruction and cumulative morbidity. Tracheobronchial involvement occurs in fewer than 5% of cases and often signals an aggressive, treatment-refractory phenotype. Despite decades of evolving endoscopic and adjuvant strategies, true long-term remission is rarely achieved. We present an extraordinary case of multifocal adult-onset RRP spanning over four decades, initially severe enough to prompt consideration for pneumonectomy, that ultimately achieved complete sustained remission following serial argon plasma coagulation (APC) and intralesional cidofovir therapy. Case Presentation A 71-year-old man, former smoker, with severe asthma, COPD, and OSA on CPAP, had a 40-year history of HPV-associated papillomatosis involving the larynx, trachea, and right lung. His disease began in 1979 with recurrent right-sided pneumonias. Bronchoscopy in 1980 revealed a papillomatous lesion in the right mainstem bronchus, and right pneumonectomy was recommended but deferred. CT chest later revealed right lower lobe bronchiectasis and a tracheal defect. Bronchoscopies from 2015 to 2016 (see images) demonstrated diffuse papillomas in the right vocal cord, trachea, and bronchi with severe dynamic collapse. Lesions were repeatedly ablated with APC and treated with intralesional cidofovir. Within three months, only a small residual lesion remained. By 2020, bronchoscopy confirmed complete clearance with no recurrence. The patient remains asymptomatic and recurrence-free to date. Discussion / Learning Points This case illustrates a rare and remarkable outcome in adult-onset RRP, with complete remission after 40 years of recurrent disease. The patient, once considered for pneumonectomy, achieved sustained control through long-term, multidisciplinary management integrating APC and cidofovir. Cidofovir, a nucleotide analogue that inhibits HPV DNA polymerase, has been shown to reduce lesion burden, prolong recurrence-free intervals, and occasionally induce remission with an excellent safety profile when injected locally. This case highlights its potential durability when combined with meticulous endoscopic therapy. Given that RRP is typically marked by relentless recurrence despite aggressive treatment, this sustained remission emphasizes that individualized, multimodal, and persistent management can sometimes overcome even the most refractory forms of this disease, offering hope for long-term control in adult tracheobronchial RRP. This abstract is funded by: None
Garcia et al. (Fri,) studied this question.