Juvenile recurrent respiratory papillomatosis treated with systemic bevacizumab: A single-center pediatric case series

BACKGROUND: Juvenile recurrent respiratory papillomatosis (JRRP) is a rare and potentially life-threatening disease caused by human papillomavirus (HPV) infection, most commonly types 6 and 11. Despite repeated surgical interventions, disease recurrence is frequent, and there is no curative treatment. Bevacizumab, a monoclonal antibody targeting vascular endothelial growth factor (VEGF), has emerged as a promising systemic therapy for severe cases. METHODS: We conducted a retrospective analysis of pediatric patients (<18 years) with JRRP who received systemic bevacizumab between 2018 and 2025 at Santa Casa de São Paulo Hospital, Brazil. Clinical, demographic, and endoscopic data were collected from electronic medical records. Bevacizumab was administered monthly (5-10 mg/kg) according to a standardized protocol, and efficacy was evaluated by comparing lesion volume before and after treatment. RESULTS: Thirteen patients were included, 69.2% male and 53.8% of African descent, with a median age at symptom onset of 18 months. All had lesions below the epiglottis, and 69.2% were tracheostomized. After 10 cycles of bevacizumab, all patients exhibited marked lesion reduction, with complete remission in 76.9%. The median number of surgical interventions decreased from 7.5 to 1.7 per year, and all tracheostomized patients were decannulated. Three patients required prolonged therapy due to persistent lesions, and no serious adverse events were observed. CONCLUSIONS: Systemic bevacizumab demonstrated significant clinical efficacy and an acceptable safety profile in children with severe or recurrent JRRP. This series supports its use as an adjuvant therapy in refractory pediatric cases. Larger multicenter studies are warranted to determine optimal treatment duration and long-term outcomes.