Long-term Outcomes of Juvenile Onset Recurrent Respiratory Papillomatosis with Pulmonary Involvement

Laryngoscope. 2021 Jul;131(7):EE2277-E2283. doi: 10.1002/lary.29376. Epub 2021 Jan 7.

Abstract

Objective: To investigate the clinical characteristics and long-term outcomes of juvenile onset recurrent respiratory papillomatosis (JORRP) with or without pulmonary involvement.

Methods: A group of patients with JORRP who had clinical course over an extended period of time (at least 5 years) in the Department of Otolaryngology Head and Neck Surgery, Beijing Tongren Hospital were included in this retrospective study. Lung/bronchus involvement was revealed by lung imaging. Data on mortality rate, frequency of surgical interventions, and age of disease onset were collected and analyzed.

Results: The 192 patients (107 male and 85 female) included had a median [quartiles] age of JORRP onset of 2 [1, 4] years, and median follow-up duration of 10 [7, 13] years; 17 patients (8.9%) had papilloma with bronchial and pulmonary involvement 7.0 [4.0, 12.5] years after the onset of the disease. Compared to patients without lung involvement, patients with lung involvement had a younger age of disease onset (P = .001), higher frequency of surgical interventions (P < .001), higher mortality rate (OR = 94.909), and an increased risk of tracheotomy that could not be decannulated (P < .001). They also had a younger age of disease onset, and a higher frequency of surgical interventions and mortality compared to patients with tracheotomy but free from lung involvement (P < .001).

Conclusions: Children with JORRP and with pulmonary involvement have a higher average number of operations per year than those without pulmonary involvement, and pulmonary involvement indicates a higher incidence of tracheotomy that cannot be decannulated.

Level of evidence: 4 Laryngoscope, 131:E2277-E2283, 2021.

Keywords: Papilloma of the respiratory tract; lung; prognosis; tracheotomy.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Age of Onset
  • Child
  • Child, Preschool
  • Female
  • Follow-Up Studies
  • Humans
  • Incidence
  • Lung / diagnostic imaging
  • Lung / pathology
  • Lung Neoplasms / diagnosis
  • Lung Neoplasms / mortality
  • Lung Neoplasms / pathology
  • Lung Neoplasms / surgery*
  • Male
  • Neoplasm Recurrence, Local
  • Papillomavirus Infections / diagnosis
  • Papillomavirus Infections / mortality
  • Papillomavirus Infections / pathology
  • Papillomavirus Infections / surgery*
  • Respiratory Tract Infections / diagnosis
  • Respiratory Tract Infections / mortality
  • Respiratory Tract Infections / pathology
  • Respiratory Tract Infections / surgery*
  • Retrospective Studies
  • Risk Factors
  • Tomography, X-Ray Computed
  • Trachea / diagnostic imaging
  • Trachea / pathology
  • Trachea / surgery
  • Tracheal Neoplasms / diagnosis
  • Tracheal Neoplasms / mortality
  • Tracheal Neoplasms / pathology
  • Tracheal Neoplasms / surgery*
  • Tracheostomy / statistics & numerical data*
  • Tracheotomy / statistics & numerical data*
  • Treatment Outcome
  • Young Adult

Supplementary concepts

  • Recurrent respiratory papillomatosis

Associated data

  • ChiCTR/ChiCTR1900027910