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Time Course of Juvenile Onset Recurrent Respiratory Papillomatosis Caused by Human Papillomavirus

Hawkes, Michael MD*; Campisi, Paolo MD†; Zafar, Rubeena MBBS*; Punthakee, Xerxes MD†; Dupuis, Annie PhD‡; Forte, Vito MD†; Ford-Jones, Elizabeth Lee MD*

Pediatric Infectious Disease Journal: February 2008 - Volume 27 - Issue 2 - pp 149-154
doi: 10.1097/INF.0b013e318159833e
Original Studies

Background: With the recent licensure of a new quadrivalent vaccine, many diseases caused by human papillomavirus (HPV) can now be prevented, including recurrent respiratory papillomatosis (RRP). The purpose of this study was to describe the burden and time course of juvenile onset RRP.

Methods: A retrospective chart review was conducted of children with airway papillomatosis at the Hospital for Sick Children in Toronto, Canada, between 1994 and 2004. Statistical methods included descriptive statistics of the cohort, a repeated events survival model, and nonlinear modeling equations to describe the time course of illness.

Results: Nine hundred twenty-six surgical procedures in 67 patients were identified through a review of surgical records. The median age at diagnosis was 3.2 years (range, 0.1–14.8 years) and the most common presenting symptom was hoarseness (75%). Adjuvant pharmacologic therapy (interferon or cidofovir) was used in 13 cases (19%). HPV types 6 or 11 were identified most commonly as the etiologic agent. Nonlinear modeling equations (exponential and quadratic) fit the observed data well, and were superior to linear models. Repeated events survival analysis identified significant prognostic variables: surgeon, adjuvant therapy, and anatomic score. A decision rule is presented that allows the time to next surgery to be predicted based on the previous surgery and the anatomic score.

Conclusions: Most patients have a decelerating rate of debulking surgeries over time, well described by our nonlinear modeling equations. Factors affecting the time course of RRP include: intersurgeon variability, the extent and severity of papillomas at the time of laryngoscopy, and the use of adjuvant medical therapies.

From the *Division of Infectious Diseases, †Department of Otolaryngology, Head and Neck Surgery, and ‡Population Health Sciences, Hospital for Sick Children, Toronto, ON, Canada.

Accepted for publication August 27, 2007.

Merck provided funding for the chart review and for presentation of results at an international conference.

Address for correspondence: Michael Hawkes, MD, Division of Infectious Diseases, Hospital for Sick Children, 555 University Ave., Toronto, ON, Canada M5G 1X8. E-mail: michael.hawkes@utoronto.ca.

© 2008 Lippincott Williams & Wilkins, Inc.