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Simple Summary Juvenile-onset recurrent respiratory papillomatosis (JoRRP) is a condition related to HPV 6 and 11 infection which is characterized by the repeated growth of benign exophytic papilloma in the respiratory tract of children. Disease progression is unpredictable leading sometimes to airway compromise and death. The aim of this study was to explore p16INK4a and expression of the RNA of HPV genes E6 and E7 with a chromogenic in situ hybridization (CISH) as biomarkers of JoRRP aggressiveness on a bicentric cohort of forty-eight children. CISH was scored semi-quantitatively as high (2+ score) and low (1+ score) levels of transcription of E6 and E7. Patients with a 2+ score had a more aggressive disease compared to those with a 1+ score. These data are a first step towards the use of biomarkers predictive of disease severity in JoRRP, this could improve the disease management, for example, by implementing adjuvant treatment at the early stages. Abstract Juvenile-onset recurrent respiratory papillomatosis (JoRRP) is a condition related to HPV 6 and 11 infection which is characterized by the repeated growth of benign exophytic papilloma in the respiratory tract. Disease progression is unpredictable: some children experience minor symptoms, while others require multiple interventions due to florid growth. The aim of this study was to explore the biomarkers of JoRRP severity on a bicentric cohort of forty-eight children. We performed a CISH on the most recent sample of papilloma with a probe targeting the mRNA of the E6 and E7 genes of HPV 6 and 11 and an immunostaining with p16INK4a antibody. For each patient HPV RNA CISH staining was assessed semi-quantitatively to define two scores: 1+, defined as a low staining extent, and 2+, defined as a high staining extent. This series contained 19 patients with a score of 1+ and 29 with a score of 2+. Patients with a score of 2+ had a median of surgical excision (SE) per year that was twice that of patients with a score of 1+ (respectively 6.1 versus 2.8, p = 0.036). We found similar results with the median number of SE the first year. Regarding p16INK4a, all patients were negative. To conclude, HPV RNA CISH might be a biomarker which is predictive of disease aggressiveness in JoRRP, and might help in patient care management.