Factors Related to Extralaryngeal Spread in Juvenile Recurrent Respiratory Papillomatosis

Abstract

Objectives/Hypothesis: To identify factors associated to increased risk of extra-laryngeal spread in pediatric patients with recurrent respiratory papillomatosis (RRP).Study Design: Retrospective chart review.Methods: A retrospective study was conducted evaluating the clinical charts of patients younger than 16 years with histopathologically confirmed RRP treated between January 2014 and December 2018. Characteristics of patients with and without extra-laryngeal disease dissemination were compared. Odds ratios were calculated and multivariate logistic regression analysis was performed.Results: Data from 82 patients were analyzed. Mean age at symptom onset was 42 months. Fifteen (18.29%) patients had extra-laryngeal spread (ELS) at time of diagnosis and in four, the disease continued to spread to other sites. Of 67 patients with disease restricted to the larynx, 17 (25.37%) developed ELS during the disease course. Human papilloma virus (HPV) typing was performed in 49 (59.8%) patients; in 28 (57.1%) HPV subtype 6 was identified and in 21 (42.9%) HPV subtype 11. ELS was found in 11 patients with serotype 11 (52.38%) and in seven patients with serotype 6 (25%) (P = .048). Statistically significant differences for ELS were also found for age at diagnosis younger than 5 years (P = .045), presence of tracheostomy (P = .031), and need for adjuvant therapy (P = .010).Conclusions: Age at diagnosis of RRP younger than 5 years and presence of tracheostomy were factors related to ELS. A statistically significant association between infection with HPV subtype 11 and ELS were also observed. Adjuvant medication might be considered a protective factor against ELS.

HPV-Associated Benign Squamous Cell Papillomas in the Upper Aero-Digestive Tract and Their Malignant Potential

“…Various hypotheses have been proposed to explain this distal spread, including the extension of papillomas by contiguity, diffuse viral contamination, and iatrogenic factors, such as laryngoscopy, bronchoscopy, tracheostomy, and surgical manipulation. Risk factors for the spread of RRP toward the lower respiratory tract include HPV11 infection, age younger than 5 years, tracheostomy performed to avoid airway obstruction, and previous invasive procedures [ 80 , 81 ]. Gélinas et al (2008) reviewed cohort studies on RRP, and 3.3% (55/1666) of the patients) had pulmonary involvement [ 82 ].…”

“…Tracheal papilloma (TP) is a distinct rarity, mostly manifested as the tracheal involvement of RRP. TPs have both a juvenile and an adult onset, presenting with either a JO- or AO-papillomatosis, mostly due to the distal spread of RRP from the larynx [ 80 , 81 , 82 ]. TPs are more common (3–26%) than pulmonary involvement (1–3%).…”

Characteristics of Recurrent Respiratory Papillomatosis and Impact Factors of Surgical Intervals

“…Previous research found that endotracheal intubation aggravated distal spread, and the longer the tube carrying time, the longer the disease time, affecting the remission rate and mortality of patients [12,13] . A study in Argentina of 82 patients with RRP under 16 years of age comparing the characteristics of RRP patients with or without extralaryngeal spread found that age younger than 5 years or history of tracheostomy at the time of diagnosis of RRP were factors associated with extralaryngeal spread, and the occurrence of extralaryngeal spread was also associated with HPV subtype 11 infection [14] .…”

The Impact of Laryngopharyngeal Reflux on Occurrence and Clinical Course of Recurrent Respiratory Papillomatosis

“…[10][11][12] The factors that act as triggers for the growth of laryngeal papillomas are still unknown, and the risk factors for the aggressive form of the disease have not yet been fully elucidated. 2,3,7,[13][14][15][16][17][18][19] Laryngopharyngeal reflux (LPR) has been proposed both as a trigger for RRP onset and as an exacerbating factor leading to disease recurrence. 12,[20][21][22][23][24] Reflux of gastric contents to the level of the larynx causes significant damage to the mucosa and has been implicated in various laryngeal pathologic lesions.…”