Long Term Outcome of Juvenile Onset Recurrent Respiratory Papillomatosis in University Kebangsaan Medical Centre

Abstract

Objective: To review the long term outcome of juvenile onset recurrent respiratory papillomatosis (JORRP) in University Kebangsaan Medical Centre (UKMMC).
Methods: A sixteen-year retrospective study – from January 2002 to June 2019 – of patients who presented with JORRP at our hospital was carried out.
Results: Nineteen cases were identified with the youngest age of presentation being eight months old. The mean follow-up time was 4.8 years. All patients had glottic papilloma, ten had multi-site involvement, namely at the subglottic, trachea, oropharynx and one case of distal spread to the lungs. The majority (71.4%) of patients with tracheostomy had extralaryngeal spread and shorter surgical intervals (a mean of 3.2 procedures per year). All our patients were treated surgically. Two patients with aggressive disease received a trial of the adjuvant therapies Gardasil and Cidofovir. However, no improvement was observed.
Conclusion: The worst prognosis is seen in younger-onset JORRP. Surgery is the mainstay of treatment and patients with aggressive disease may need adjuvant therapy. Unfortunately, there is a lack of a standard protocol for adjuvant therapies. Tracheostomy increases the risk of extralaryngeal spread and aggressive disease, and thus should be reserved for patients with severe airway compromise.

Keywords

• Human papillomavirus infection,respiratory tract,pediatrics,tracheostomy

References

1. 1. Fasunla AJ, Lasisi OA. Diagnostic challenges of laryngeal papillomatosis and its implications among children in developing country. Int J Pediatr Otorhinolaryngol 2009;73:593-5.
2. 2. Derkay CS, Wiatrak B. Recurrent respiratory papillomatosis: a review. Laryngoscope. 2008;118:1236-47.
3. 3. Gandhi S, Jacob R. Remission in juvenile-onset recurrent respiratory papillomatosis. J Laryngol Voice 2012;2:30-4.
4. 4. Ivancic R, Iqbal H, deSilva B, Pan Q, Matrka L. Current and future management of recurrent respiratory papillomatosis. Laryngoscope Investig Otolaryngol 2018;3:22-34.
5. 5. Lee SM, Park JS, Norwitz ER, et al. Risk of vertical transmission of human papillomavirus throughout pregnancy: a prospective study. PLoS One 2013;8:e66368.
6. 6. Winckworth LC, Nichol R. Question 2: do caesarean sections reduce the maternal-fetal transmission rate of human papillomavirus infection? Arch Dis Child 2010;95:70-3.
7. 7. Omland T, Akre H, Lie KA, Jebsen P, Sandvik L, Brøndbo K. Risk factors for aggressive recurrent respiratory papillomatosis in adults and juveniles. PLoS One 2014;9:e113584.
8. 8. Long YT, Sani A. Recurrent respiratory papillomatosis. Asian J Surg 2003;26:112-6.

9. 9. Aaltonen LM, Rihkanen H, Vaheri A. Human papillomavirus in larynx. Laryngoscope 2002;112:700-7.
10. 10. Schraff S, Derkay CS, Burke B, Lawson L. American Society of Pediatric Otolaryngology members’ experience with recurrent respiratory papillomatosis and the use of adjuvant therapy. Arch Otolaryngol Head Neck Surg 2004;130:1039-42.
11. 11. Murono S, Nakanishi Y, Tsuji A, et al. Intralesional cidofovir injection for recurrent respiratory papillomatosis in Japan. Auris Nasus Larynx 2016;43:541-5.
12. 12. Pransky SM, Albright JT, Magit AE. Long-term follow-up of pediatric recurrent respiratory papillomatosis managed with intralesional cidofovir. Laryngoscope 2003;113:1583-7.
13. 13. Rogers DJ, Ojha S, Maurer R, Hartnick CJ. Use of adjuvant intralesional bevacizumab for aggressive respiratory papillomatosis in children. JAMA Otolaryngol Head Neck Surg 2013;139:496-501.
14. 14. Kin Cho Goon P, Scholtz LU, Sudhoff H. Recurrent respiratory papillomatosis (RRP)-time for a reckoning? Laryngoscope Investig Otolaryngol 2017;2:184-6.
15. 15. Hermann JS, Weckx LY, Monteiro Nürmberger J, Santos Junior GF, Campos Pignatari AC, Nagata Pignatari SS. Effectiveness of the human papillomavirus (types 6, 11, 16, and 18) vaccine in the treatment of children with recurrent respiratory papillomatosis. Int J Pediatr Otorhinolaryngol 2016;83:94-8.
16. 16. Novakovic D, Cheng ATL, Zurynski Y, et al. A prospective study of the incidence of juvenile-onset recurrent respiratory papillomatosis after implementation of a national HPV vaccination program. J Infect Dis 2018;217:208-12.