SCIENTIFIC
Systemic Cidofovir as a Treatment for Recurrent Respiratory Papillomatosis in a Child
AUTHORS: Laurel Bolton, DO 1
Umer Muhammad, MD 1 John Bishara, MD 1 Marcus Shaffer, MD 1 Raheel Khan, MD 1 Mohamad Badawi, MD 1
1
Charleston Area Medical Center, West Virginia University, Charleston, WV
CORRESPONDING AUTHOR: Mohamad Badawi, MD mohamad. badawi @ camc. org
DISCLOSURES: None
ETHICS STATEMENT: Permission obtained from parent.
FUNDING: None
CASE PRESENTATION
A 5-year-old boy presented with tracheostomy dependence from severe laryngeal obstruction from recurrent respiratory papillomatosis( RRP). He was born prematurely at 31 weeks gestational age due to prolonged premature rupture of membranes. The pregnancy was complicated by maternal hepatitis C and in-utero exposure to nicotine, buprenorphine, and marijuana. At 7 months of age, the boy presented with progressive respiratory distress and stridor and was treated in our pediatric intensive care unit. A flexible bronchoscopy noted that a mass-like structure was obstructing the vocal cords. Due to severe laryngeal obstruction, the procedure was stopped. He was transferred to a tertiary children’ s hospital and diagnosed with severe RRP involving the true vocal cords. A tracheostomy was placed due to severe airway obstruction. Over the following two years, he underwent debulk-
ABSTRACT
Recurrent respiratory papillomatosis is a rare but devastating disease. It is often characterized by numerous recurrences that are often refractory to surgical and adjunctive treatments. We present the case of a 5-year-old male with tracheostomy dependence from
ing procedures every four weeks. Both intralesional and systemic bevacizumab were initiated in another tertiary care center. After a short interval of stability, his disease progressed to the carina. At 2 years and 9 months, he required emergent debridement for glottic and subglottic obstruction from the papillomas. The anterior vocal folds had scarred together from multiple debridement procedures. There was also complete obstruction of the trachea by papillomas distal to the tracheostomy tube. After debridement, there was a clear visualization of the carina.
As the bevacizumab effect was suboptimal, the patient continued with scheduled debulking procedures until 3 years and 5 months of age( Figure 1), when he began cidofovir infusions at our institution. Cidofovir was prescribed as outlined in the work of Valckenborgh et al, which consisted of cidofovir( 5 mg / kg) over one hour weekly for four weeks followed by every other week for eight weeks, then every third week for nine weeks, and finally every four weeks for 20 more weeks. The total duration of treatment was one year. We ensured proper hydration, along with oral probenecid, to reduce nephrotoxicity. Serum electrolytes and renal function were closely monitored throughout the treatment.
One month after starting systemic cidofovir, there was significant improvement in the child’ s RRP. The papillomas were still present in the distal trachea but less dense before the start of cidofovir treatments.
Our patient tolerated the cidofovir infusion well without adverse effects. The debridement frequency significantly decreased
recurrent respiratory papillomatosis with distal seeding who was successfully treated with systemic cidofovir and palliative debulking procedures after failing treatment with intralesional and systemic bevacizumab.
FIGURE 1
Fiberoptic scope of vocal cords at the start of cidofovir treatment.
FIGURE 2
Fiberoptic scope of vocal cords at the end of cidofovir treatment.
FIGURE 3
Fiberoptic scope of vocal cords at the end of cidofovir treatment.
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