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Successful Treatment of Juvenile Laryngeal Papillomatosis-Related Multicystic Lung Disease With Cidofovir^* Case Report and Review of the Literature

David R. Dancey, MD; Dean W. Chamberlain, MD; Mel Krajden, MD; Joel Palefsky, MD; P.W. Alberti, MD and Gregory P. Downey, MD, FCCP

^* From the Division of Respirology (Drs. Dancey and Downey) and the Division of Medical Microbiology (Dr. Krajden), Department of Medicine, the Department of Laboratory Medicine and Pathobiology (Dr. Chamberlain), and the Division of Otolaryngology (Dr. Alberti), Department of Surgery, University of Toronto, Ontario, Canada; and the Department of Laboratory Medicine (Dr. Palefsky), University of California, San Francisco, CA. Current address: Provincial Lab, British Columbia Centre for Disease Control, Vancouver, British Colombia, Canada.

Correspondence to: Gregory P. Downey, MD, FCCP, EN 10–212, The Toronto General Hospital, 200 Elizabeth St, Toronto, Ontario, Canada M5G 2C4; e-mail: gregory.downey{at}utoronto.ca

Cidofovir, a nucleoside analog antiviral agent, has been used with moderate success in the treatment of juvenile laryngeal papillomatosis (JLP) by direct intralesional injection. We report the first case where IV cidofovir was used successfully to treat a rare but lethal multicystic lung disease complicating JLP. A 35-year-old woman with a history of JLP requiring multiple laser ablations of laryngeal papillomata each year presented with hemoptysis and was found on CT scan to have bilateral, multiple pulmonary nodules and cysts. The results of BAL fluid analysis demonstrated no evidence of malignancy, and cultures were negative for fungi and mycobacteria. Molecular DNA typing of a biopsy specimen obtained from a laryngeal papilloma confirmed infection with human papilloma virus type 11. She received 12 months of treatment with IV cidofovir followed by 9 months of combined treatmentwith IV cidofovir and subcutaneous interferon--2A. This therapeutic regime resulted in a markedly decreased requirement for surgical removal of laryngeal papillomata, and CT scanning documented the regression of the lesions in the lung parenchyma that persisted after the discontinuation of therapy. The results of this case demonstrate that cidofovir may be used successfully to treat JLP-related lung disease and suggest that further studies are warranted.

Key Words: cidofovir • human papilloma virus • interferon • juvenile laryngeal papillomatosis

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