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Intralesional cidofovir for pediatric recurrent respiratory papillomatosis
David L Mandell1, Ellis M Arjmand, David J Kay
1Department of Pediatric Otolaryngology, Children's Hospital of Pittsburgh, University of Pittsburgh Voice Center, University of Pittsburgh, Pittsburgh, PA, USA. david.mandell@chp.edu
Insights
Combined surgical debulking and intralesional cidofovir injections showed improved disease response in children with recurrent respiratory papillomatosis (RRP). This treatment appears safe and effective for pediatric RRP management.
Area of Science:
- Otolaryngology
- Pediatric Pulmonology
- Viral Diseases
Background:
- Recurrent respiratory papillomatosis (RRP) is a rare, chronic disease caused by human papillomavirus (HPV).
- RRP can lead to significant airway obstruction and morbidity in children.
- Current treatment primarily involves surgical debulking, which is often insufficient for long-term disease control.
Purpose of the Study:
- To compare the efficacy of combined surgical debulking and intralesional cidofovir injections versus surgical debulking alone in pediatric patients with RRP.
- To evaluate the safety and disease response in children with RRP treated with intralesional cidofovir.
Main Methods:
- Retrospective medical record review of seven children with RRP at a tertiary care children's hospital.
- Four subjects received combined surgical debulking and intralesional cidofovir injections every two months.
- Three control subjects received repeated surgical debulking only. Disease severity was assessed using intraoperative endoscopic photographs and papilloma staging scores.
Main Results:
- The cidofovir group demonstrated significantly lower final papilloma staging scores compared to the control group (P < .05).
- No significant differences were observed in within-group scores between initial and final assessments for either the cidofovir or control groups.
- The treatment with intralesional cidofovir was found to be safe in the studied pediatric population.
Conclusions:
- Intralesional cidofovir injection, combined with surgical debulking, represents a safe and effective treatment option for pediatric recurrent respiratory papillomatosis.
- Wider consideration of cidofovir for pediatric RRP treatment is warranted.
- Larger studies are needed to definitively establish the impact of cidofovir on RRP management.
Objective:
To compare disease response among children with recurrent respiratory papillomatosis (RRP) who underwent combined surgical debulking and intralesional cidofovir injections vs repeated surgical debulking only.
Design:
Retrospective medical record review; follow-up range: 16 to 56 months.
Setting:
Tertiary care children's hospital.
Patients:
Seven children with RRP, including 4 subjects treated with cidofovir and 3 controls matched for age and initial papilloma staging score.
Interventions:
Subjects treated with cidofovir underwent combined surgical debulking and intralesional cidofovir injection every 2 months until disease remission. Control subjects underwent repeated surgical debulking at individually determined intervals.
Main Outcome Measures:
Intraoperative endoscopic photographs were retrospectively assigned papilloma staging scores. Cidofovir and control group comparisons were made using nonparametric 2-sample Wilcoxon rank-sum (Mann-Whitney) testing, and comparisons of initial and final papilloma staging scores were made using nonparametric matched-pair Wilcoxon signed-rank testing.
Results:
The final cidofovir group scores were significantly lower than the control group scores (P < .05). Within-group differences between initial and final scores were not significant (cidofovir group, P = .07; control group, P = .29).
Conclusions:
Four children with RRP were safely and successfully treated with intralesional cidofovir injection. Consideration should be given to using cidofovir more widely for treatment of pediatric RRP. Larger numbers in the cidofovir and control groups are needed in future studies to determine the true impact of cidofovir on management of this disease.
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