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Published on: March 8, 2012
Inhaled cidofovir as an adjuvant therapy for recurrent respiratory papillomatosis
Jeffrey Ksiazek1, Jeremy D Prager, Gordon H Sun
1Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.
Insights
Inhaled cidofovir shows promise for treating severe recurrent respiratory papillomatosis in infants when conventional therapies fail. This novel inhaled treatment significantly reduced papilloma burden in a pediatric case study.
Area of Science:
- Pediatric Otolaryngology
- Viral Infections
- Respiratory 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, particularly in infants.
- Conventional treatments include surgical debridement and various medical therapies, often with limited success in severe cases.
Observation:
- A 4-month-old infant with a progressively worsening airway obstruction due to extensive glottic papillomas presented with respiratory distress.
- Despite multiple microdebridements, intralesional cidofovir, and systemic interferon-alpha, the infant's condition deteriorated, requiring prolonged hospitalization.
- Conventional treatments failed to control the disseminated papillomatous disease.
Findings:
- Inhaled cidofovir therapy (40 mg daily, 12 days on/2 days off) was initiated as a last resort.
- Within 6 weeks, inhaled cidofovir led to substantial improvement in the papillomatous disease burden.
- The patient's condition significantly improved, with a reduction in the Derkay score from 40 to 23.
Implications:
- Inhaled cidofovir may represent a novel and effective therapeutic option for severe, treatment-refractory recurrent respiratory papillomatosis in pediatric patients.
- This approach warrants further investigation to establish its long-term efficacy and safety profile.
- The findings suggest a potential paradigm shift in managing aggressive RRP cases unresponsive to standard treatments.
Abstract:
A previously healthy, full-term, 4-month-old boy presented with progressively weakening cry, hoarseness, and increased work of breathing. Flexible fiberoptic laryngoscopy revealed glottic papillomas, which were endoscopically removed with a microdebrider in the operating room (Derkay score 23). The patient was diagnosed with recurrent respiratory papillomatosis that disseminated throughout his airway. Despite biweekly serial microdebridements, intralesional cidofovir, and systemic interferon-α, the patient's health declined substantially (Derkay score 40), culminating in a 47-day hospitalization due to complications of his disease. Inhaled cidofovir was initiated after all conventional treatments had failed. Within 6 weeks of therapy (40 mg daily per treatment, 12 days on and 2 days off), the papillomatous disease improved substantially (Derkay score 23). While inhaled cidofovir appeared to significantly reduce papillomatous disease burden in this patient, further investigation into its long-term effectiveness and safety profile is necessary.
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