Journal of voice : official journal of the Voice Foundation · 2026
Efficacy of Systemic Bevacizumab in Juvenile-Onset Recurrent Respiratory Papillomatosis: An Individual Patient Data Meta-Analysis.
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Hortobagyi David, Denk-Linnert Doris-Maria, Altmann Marie, Valentinuzzi Katharina, Grossmann Tanja, Kaider Alexandra, Kranebitter Veronika
Аннотация
OBJECTIVE: Juvenile-onset recurrent respiratory papillomatosis (joRRP) is a benign but often aggressive HPV-related disease of the pediatric airway, frequently requiring repeated surgical debulking. These repeated interventions are associated with substantial morbidity and impaired quality of life, particularly due to persistent dysphonia. Systemic bevacizumab, an anti-VEGF monoclonal antibody, has emerged as a promising adjuvant therapy, but individual patient-level evidence synthesizing its efficacy across studies is lacking.
METHODS: We performed an individual patient data meta-analysis to evaluate the effect of systemic bevacizumab on surgical intervention rates in patients with joRRP. Following a systematic search of four electronic databases, six studies comprising 33 patients met eligibility criteria. Thirty patients with documented follow-up duration were included in the primary analysis. Surgical rates before and after bevacizumab initiation were compared using a generalized linear mixed model with a negative binomial distribution and log link, with follow-up duration as an offset. Changes in Derkay scores were assessed using the paired Wilcoxon signed-rank test.
RESUTS: Prior to treatment, patients had undergone a median of 16 surgical interventions (IQR 5-32). The estimated surgical rate decreased from 38.6 to 1.8 procedures per 100 patient-months after bevacizumab initiation, corresponding to a rate ratio of 0.047 (95% CI 0.017-0.128) and a relative reduction of 95.3% (P < 0.0001). Among nine patients with paired Derkay scores, the median score decreased from 8 (IQR 5-15) to 0 (IQR 0-1; P = 0.0039). Adverse events were mild, most commonly epistaxis and proteinuria, with no serious adverse events reported.
CONCLUSIONS: This meta-analysis suggests that systemic bevacizumab may substantially and safely reduce surgical burden in pediatric patients with aggressive joRRP.
METHODS: We performed an individual patient data meta-analysis to evaluate the effect of systemic bevacizumab on surgical intervention rates in patients with joRRP. Following a systematic search of four electronic databases, six studies comprising 33 patients met eligibility criteria. Thirty patients with documented follow-up duration were included in the primary analysis. Surgical rates before and after bevacizumab initiation were compared using a generalized linear mixed model with a negative binomial distribution and log link, with follow-up duration as an offset. Changes in Derkay scores were assessed using the paired Wilcoxon signed-rank test.
RESUTS: Prior to treatment, patients had undergone a median of 16 surgical interventions (IQR 5-32). The estimated surgical rate decreased from 38.6 to 1.8 procedures per 100 patient-months after bevacizumab initiation, corresponding to a rate ratio of 0.047 (95% CI 0.017-0.128) and a relative reduction of 95.3% (P < 0.0001). Among nine patients with paired Derkay scores, the median score decreased from 8 (IQR 5-15) to 0 (IQR 0-1; P = 0.0039). Adverse events were mild, most commonly epistaxis and proteinuria, with no serious adverse events reported.
CONCLUSIONS: This meta-analysis suggests that systemic bevacizumab may substantially and safely reduce surgical burden in pediatric patients with aggressive joRRP.