Inhaled Cidofovir as an Adjuvant Therapy for Recurrent Respiratory Papillomatosis

Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.
Otolaryngology Head and Neck Surgery (Impact Factor: 2.02). 04/2011; 144(4):639-41. DOI: 10.1177/0194599810395353
Source: PubMed


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.

14 Reads
  • [Show abstract] [Hide abstract]
    ABSTRACT: Recurrent respiratory papillomatosis (RRP) is a rare, benign disease with no known cure. RRP is caused by infection of the upper aerodigestive tract with the human papillomavirus (HPV). Passage through the birth canal is thought to be the initial transmission event, but infection may occur in utero. HPV vaccines have helped to provide protection from cervical cancer; however, their role in the prevention of RRP is undetermined. Clinical presentation of initial symptoms of RRP may be subtle. RRP course varies, and current management focuses on surgical debulking of papillomatous lesions with or without concurrent adjuvant therapy.
    No preview · Article · Jun 2012 · Otolaryngologic Clinics of North America
  • [Show abstract] [Hide abstract]
    ABSTRACT: Juvenile-onset recurrent respiratory papillomatosis is a devastating illness of children who develop obstruction of the airway from neoplastic growths of infectious origin. The disease is caused by human papillomavirus (HPVs), types 6 and 11, incorporating itself into the genome of the respiratory mucosa. The consequence of this genetic piracy is a morphologic change from an airway lining that is flat and smooth to an irregular “mulberry like” hyperplasia. The growths seem to have a predilection for the glottic mucous membranes but can involve any part of the aerodigestive tract. The most common symptom is hoarseness, and children can have an alarming degree of obstruction with this as their only symptom. When hoarseness progresses to stridor, respiratory distress is impending. The dynamics of disease recurrence vary widely, with the more severe, rapidly recurring disease requiring frequent operative excisions to maintain airway patency. Surgery has been the mainstay of management but is not curative. Adjuvant therapies are elusively effective, unpredictably decreasing the disease activity and increasing the duration between surgeries. The quadrivalent HPV vaccine (Gardisil; Merck, NJ) is promising to reduce the HPV infection rate in young women, with the hope that by eliminating this reservoir of disease, the incidence of HPV-related illnesses, juvenile-onset including recurrent respiratory papillomatosis, will dramatically drop.
    No preview · Article · Jun 2012 · Operative Techniques in Otolaryngology-Head and Neck Surgery
  • [Show abstract] [Hide abstract]
    ABSTRACT: Cidofovir has shown antiproliferative effects against human papillomavirus (HPV)-positive cells and successfully suppressed the growth of HPV-positive xenografts in athymic nude mice. The present study evaluated the effect of cidofovir on several disease parameters in this animal model. Intratumoral administration of cidofovir resulted in a beneficial effect on body weight gain, a reduction in splenomegaly, a partial restoration of tryptophan catabolism, and diminished the inflammatory state induced by the xenografts. Administration of cidofovir to tumor-free animals did not have a direct effect on these parameters. Beyond suppressing tumor growth, intratumoral treatment with cidofovir ameliorated the pathology associated with HPV-tumor growth.
    No preview · Article · Nov 2012 · Cancer letters
Show more