Clinical implications of alpha, beta, and gamma HPV infection in juvenile onset recurrent respiratory papillomatosis

Martina Bertinazzi,Marta Sbaraglia,Cesare Cutrone,Marianna Sari,Piero Nicolai,Jerry Polesel,Massimo Tommasino,Tarik Gheit,Sandrine Mckay-Chopin,Angelo Paolo Dei Tos,Paolo Boscolo-Rizzo

doi:10.1007/s00405-021-07040-9

Abstract

PurposeThe aim of our study was to evaluate the prevalence of different HPV genera—alpha, beta and gamma—in Juvenile onset Recurrent Respiratory Papillomatosis (JoRRP) and examine the association of type and genus-specific viral features with the clinical outcome of disease.MethodsThis retrospective observational study included consecutive patients with JoRRP who were treated in a referral centre between October 2000 and October 2020. All patients underwent cold excision and laser vaporisation of papillomatous lesions. Samples were analysed for the presence of 120 viral genotypes (22 alpha-HPV, 46 beta-HPV, 52 gamma-HPV) using a highly sensitive multiplex genotyping assay.ResultsTwenty patients with JoRRP, aged 0.3–11 years, were included, with a median follow-up of 13.5 years. All samples were HPV DNA positive: 20 (100%) for alpha-HPV DNA; 7 (35%) for beta—HPV DNA; 0 for gamma-HPV DNA. Three groups were defined according to the number of infections: seven cases (35%) with HPV mono-infection; ten cases (50%) with HPV double-infection; three cases (15%) with ≥ 3 HPV infections. At diagnosis, patients with ≥ 3 HPV infections reported higher median Derkay’s score than those with mono-infection (21 vs 14, P = 0.018). Number of HPV infections was also associated with clinical outcomes, with an average of 0.5 surgical procedures/year in patients with mono-infection, 1.2 for double-infection, 2.6 for ≥ 3 infections (P = 0.006).ConclusionDespite the small sample size, these preliminary data support an association between the number of different alpha and beta HPV co-infections and the clinical severity of the disease.

Highlights

Juvenile onset recurrent respiratory papillomatosis (JoRRP) is a rare and challenging HPV-induced disease, frequently associated with mucosal HPV-type infection, in particular HPV 6 and 11, and characterised by the growth of squamous papillomata in the airway epithelium [1]
Many studies have investigated whether the severity and aggressivity of JoRRP may be influenced by the infection with mucosal HPV types, 6 and 11, but the results provided inconsistent and contrasting conclusions [1, 6]
The data show that all JoRRP were associated with infections of mucosal alpha HPV types 6 and 11, which are well-known etiological factors of JoRRP

Summary

Introduction

Juvenile onset recurrent respiratory papillomatosis (JoRRP) is a rare and challenging HPV-induced disease, frequently associated with mucosal HPV-type infection, in particular HPV 6 and 11, and characterised by the growth of squamous papillomata in the airway epithelium [1]. It is defined by the onset before 12 years of age with the peak of incidence being commonly before 5 years [2]. The new adjuvant therapies appear to be promising in improving the control of the disease [5] It could be, interesting and innovating having biological

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