C67-20 A Case of Recurrent Respiratory Papillomatosis Treated With Bevacizumab and Nivolumab

Abstract A 24-year-old man was diagnosed at age 4 with recurrent respiratory papillomatosis (RRP), +HPV, low-risk category. He has had repeated laryngeal and tracheal debridement of airway papillomas. He has also had recurrent hemoptysis. Chest imaging has shown multiple solid and cavitating nodules, bronchiectasis, and thin-walled cysts. He was initially treated with 20 cycles of bevacizumab, with a marked improvement in his laryngeal disease. However, his pulmonary disease continued to progress. In May 2020, bevacizumab was discontinued, and he was started on nivolumab based on a published case series in refractory RRP. Although there was radiologic evidence of improvement in his pulmonary disease, his laryngeal papillomatosis worsened over 4 months of nivolumab therapy. In September 2020, in an effort to control both his pulmonary and upper airway disease, the patient was started on combination therapy with nivolumab and bevacizumab every 3 weeks. His disease remained stable for almost 2 years, until he was hospitalized for an intracranial abscess. His RRP treatment was held for several months. In February 2023, he was admitted for hemoptysis and ultimately underwent lobectomy for a cavitating lesion in the RLL. He remained off any therapy for over a year with close surveillance. During this period, he had increased growth of his laryngeal papillomas and was restarted on bevacizumab. Once again, his upper airway disease responded well, but he had progression of his pulmonary disease. He is currently on bevacizumab and nivolumab every 2 weeks. His laryngeal disease remains under control, with a mixed response in his chest. RRP is a disease caused by chronic infection of HPV-6 and HPV-11 with predilection for the respiratory epithelium. In the 8.9% of RRP patients with pulmonary involvement, there is a significant increase in morbidity as well as lifetime risk of malignant transformation. Small trials have shown positive results with bevacizumab therapy in patients with RRP, including those with pulmonary involvement. However, there are no available systemic treatments for refractory disease. A previous case series of 2 patients with refractory RRP demonstrated some clinical benefit of nivolumab. The decision to trial combination therapy with a VEGF inhibitor and PD-1 blocker in this patient was based on a Phase II trial suggesting clinical efficacy in relapsed ovarian cancer. To our knowledge, this is the first report of combined bevacizumab and nivolumab for RRP. While this patient’s pulmonary disease showed some response to therapy, clearly, more clinical trials are needed. This abstract is funded by: None