Background Recurrent Respiratory Papillomatosis (RRP) is a rare disease characterized by the growth of papillomas in the airway and especially the larynx. The clinical course is highly variable among individuals and there is poor understanding of the factors that drive an aggressive vs an indolent course. Methods A convenience cohort of 339 affected subjects with papillomas positive for only HPV6 or HPV11 and clinical course data available for 1 year or more, from a large multicenter international study were included. Exploratory data analysis was conducted followed by inferential analyses with frequentist and Bayesian statistics. Results We examined 339 subjects: 82% were diagnosed prior to the age of 18 years, 65% were infected with HPV6, and 69% had an aggressive clinical course. When comparing age at diagnosis with clinical course, the probability of aggressiveness is high for children under five years of age then drops rapidly. For patients diagnosed after the age of 10 years, an indolent course is more common. After accounting for confounding between HPV11 and young age, HPV type was minimally associated with aggressiveness. Fast and Frugal Trees (FFTs) were utilized to determine which algorithms yield the highest accuracy to classify patients as having an indolent or aggressive clinical course and consistently created a branch for diagnostic age at ~5 years old. There was no reliable strong association between clinical course and socioeconomic or parental factors. Conclusion In the largest cohort of its type, we have identified a critical age at diagnosis which demarcates a more aggressive from less aggressive clinical course.
BackgroundRecurrent respiratory papillomatosis (RRP) is characterized by repeated formation of papillomas in the respiratory tract and is caused by human papillomavirus (HPV) types 6 and 11. Women with genital HPV infection are slow to develop weak humoral immunity, but respond robustly to the HPV vaccine. We wondered if people with RRP had a similar immune response. MethodsA convenience cross-sectional sample of patients with RRP were recruited into one of four groups: 1) adults and adolescents with active RRP, 2) children with active RRP, 3) RRP patients who had undergone HPV vaccination prior to enrollment and, 4) people with RRP who were in remission. Anti-HPV6 and HPV11 serology was determined by cLIA on a single blood draw. OPEN ACCESS Citation: Buchinsky FJ, Ruszkay N, Valentino W, Derkay CS, McClay JE, Bastian RW, et al. (2020) In RRP, serologic response to HPV is frequently absent and slow to develop. PLoS ONE 15(3): e0230106. https://doi. ConclusionPeople with RRP are capable of developing a humoral response to HPV6 and HPV11. That response appears to be robust when initiated by the HPV vaccine, but either nonexistent or slow to develop in response to infection. Most in remission do not have demonstrable antibody levels against HPV6 or HPV11.
ObjectiveThe Clinical Assessment Score‐15 (CAS‐15) has been validated as an office‐based assessment for pediatric sleep‐disordered breathing in otherwise healthy children. Our objective was to determine the generalizability of the CAS‐15 in a multi‐institutional fashion.MethodsFive hundred and thirty children from 13 sites with suspected sleep‐disordered breathing were recruited, and the investigators completed the CAS‐15. Based on decisions made in the course of clinical care, investigators recommended overnight polysomnography, observation, medical therapy, and/or surgery. Two hundred and forty‐seven subjects had a follow‐up CAS‐15.ResultsMean age was 5.1 (2.6) years; 54.2% were male; 39.1% were white; and 37.0% were African American. Initial mean (standard deviation [SD]) CAS‐15 was 37.3 (12.7), n = 508. Spearman correlation between the initial CAS‐15 and the initial apnea‐hypopnea index (AHI) was 0.41 (95% confidence interval [CI], 0.29, 0.51), n = 212, P < .001. A receiver‐operating characteristic curve predicting positive polysomnography (AHI > 2) had an area under the curve of 0.71 (95% CI, 0.63, 0.80). A score ≥ 32 had a sensitivity of 69.0% (95% CI, 61.7, 75.5), a specificity of 63.4% (95% CI, 47.9, 76.6), a positive predictive value of 88.7% (95% CI, 82.1, 93.1), and a negative predictive value of 32.9% (95% CI, 23.5, 44.0) in predicting positive polysomnography. Among children who underwent surgery, the mean change (SD) score was 30.5 (12.6), n = 201, t = 36.85, P < .001, effect size = 3.1.ConclusionThis study establishes the generalizability of the CAS‐15 as a useful office tool for the evaluation of pediatric sleep‐disordered breathing.Level of Evidence2B Laryngoscope, 130:2256–2262, 2020
Powered intracapsular tonsillectomy and adenoidectomy improved OSA in this series of pediatric patients by reducing obstructive apneas and hypopneas, oxygen desaturation, arousal index, carbon dioxide level, and snoring, as well as increasing oxygen saturation nadir. Results are comparable to those described for traditional electrocautery tonsillectomy and support the use of PITA for the treatment of severe OSA in children with adenotonsillar hypertrophy.
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