Intersurgical interval increased with use of quadrivalent human papillomavirus vaccine (Gardasil) in a pediatric patient with recurrent respiratory papillomatosis: A case report


This is a case of a 4 year old female with recalcitrant recurrent respiratory papillomatosis with decreasing intersurgical interval that had improvement in clinical course after administration of the quadrivalent HPV vaccine.


Recurrent respiratory papillomatosis (RRP) is an unpredictable, benign upper aerodigestive tract infection, most commonly effecting the larynx with an approximate incidence of 4.3 per 100,000 people in the United States [1]. There is a bimodal age distribution whereas patients present in either early childhood or adulthood [2]. If RRP is diagnosed in early childhood it has shown to take on a more aggressive and recurring course of disease. Due to the laryngeal involvement, hoarseness is the first clinically apparent symptom with other findings localizing the lesion to the upper airwayddyspnea, stridor, recurrent upper respiratory infections, pneumonia, dysphagia, chronic cough, and/or failure to thrive [1]. Although benign in nature, RRP, which has no known cure, has shown to have significant morbidity due to the multiple surgeries. Thus, adjuvant therapies have been sought to increase the intersurgical interval and decrease the overall surgeries undergone by patients. Human papillomavirus (HPV) types 6 and 11 account for most cases of RRP, with HPV-11 being the most common and aggressive type. Due to these findings, the quadrivalent HPV vaccine (Gardasil) has been examined in adjuvant therapies with the standard of care, surgical management. 2.

Case report

A 4 year old female with RRP due to HPV type 11, requiring multiple surgical interventions, subsequently received the quadrivalent human papillomavirus (HPV) vaccine (Gardasil) with a positive clinical response. She was born to a G4P3 mother HPVþ mother with and delivered vaginally while mother had active papilloma. She presented with biphasic stridor at 12 months of age found to have obstructing papilloma. The patient was given bevacizumab injections to the surgical site three times in total with doses varying from 1 to 3 mg, but her clinical course continued to deteriorate with decreasing symptom free intervals. She had a total of 13 surgeries with intersurgical interval of 1.9 months with use of a microdebrider and bevacizumab as the surgical modality and adjuvant therapy, respectively. She presented at three years of age to the emergency department one month following debridement with biphasic stridor (Fig. 1). After subsequent debridement, the choice was given to parents to pursue other adjuvant therapy including possible systemic treatment or vaccine due to concern for increasing airway obstruction prior to surgical intervention and less clinical response following her surgical intervention and bevacizumab injection. Written informed consent was obtained from parents regarding the off-label use of the Gardasil vaccine in consent process developed by Nationwide Children’s Hospital. Three injections were given intramuscularly with the second injection occurring 2 months after initiating the treatment and the final vaccination given 6 months later. The vaccine was given in conjunction with surgical intervention, and she received one dose of bevacizumab during the period of vaccination. Following initiation of the vaccine, the intersurgical interval increased from 1.9 months to 3.5 months. Upon completion of the vaccine course, she has currently had a symptom free interval of 8 months, which is her longest symptom free duration. More importantly, her symptoms and voice quality have improved immensely following use of the vaccine. In addition, the average Derkay Severity Score prior to vaccination was 12. The three surgeries following initiation of vaccination, the average score has been 4. Post-vaccination titers of HPV antibodies have not been obtained (Fig. 2).