Advocacy for Recurrent Respiratory Papillomatosis

Juvenile-onset recurrent respiratory papillomatosis (JoRRP) is the most common benign neoplasm of the larynx in children, presenting with significant variation in clinical course and potential for progression to malignancy. Since JoRRP is driven by human papillomavirus (HPV), we evaluated viral factors in a prospective cohort to identify predictive factors of disease severity. Twenty children with JoRRP undergoing routine debridement of papillomas were recruited and followed for ≥1 year. Demographical features, clinical severity scores, and surgeries over time were tabulated. Biopsies were used to establish a tissue bank and primary cell cultures for HPV6 vs. HPV11 genotyping and evaluation of viral gene expression. We found that patients with HPV11+ disease had an earlier age at disease onset, higher frequency of surgeries, increased number of lifetime surgeries, and were more likely to progress to malignancy. However, the amplitude of viral E6/E7 gene expression did not account for increased disease severity in HPV11+ patients. Determination of HPV strain is not routinely performed in the standard of care for JoRRP patients; we demonstrate the utility and feasibility of HPV genotyping using RNA-ISH for screening of HPV11+ disease as a biomarker for disease severity and progression in JoRRP patients.

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The Impact of Laryngopharyngeal Reflux on Occurrence and Clinical Course of Recurrent Respiratory Papillomatosis

The Laryngoscope ◽

10.1002/lary.29793 ◽

2021 ◽

Author(s):

Daša Gluvajić ◽

Maja Šereg‐Bahar ◽

Aleš Jerin ◽

Rado Janša ◽

Irena Hočevar‐Boltežar

Keyword(s):

Clinical Course ◽

Laryngopharyngeal Reflux ◽

Recurrent Respiratory Papillomatosis ◽

The Impact

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Pembrolizumab Induced Acute Persistent Airway Disease in a Patient with Recurrent Respiratory Papillomatosis (RRP)

Annals of Otology Rhinology & Laryngology ◽

10.1177/00034894211021276 ◽

2021 ◽

pp. 000348942110212

Author(s):

Kathryn Marcus ◽

Daniel J. Lee ◽

Jeffrey S. Wilson ◽

Richard J. H. Smith ◽

Michael Puricelli

Keyword(s):

Respiratory Failure ◽

Inhaled Corticosteroids ◽

Checkpoint Inhibitor ◽

Ventilatory Support ◽

Airway Disease ◽

Recurrent Respiratory Papillomatosis ◽

Pulmonary Function Testing ◽

Hypoxic Respiratory Failure ◽

Checkpoint Inhibitor Therapy ◽

Function Testing

Objective: To present an uncommon but serious, recently identified complication of checkpoint inhibitor therapy in a patient treated with pembrolizumab infusion for disseminated recurrent respiratory papillomatosis (RRP). Methods: Case report. Results: A 43-year-old woman with underlying asthma developed acute hypoxic respiratory failure within 24 hours of her third infusion of pembrolizumab for treatment of intractable, disseminated recurrent respiratory papillomatosis. Pulmonary function testing revealed a severe intra-thoracic obstructive ventilatory defect. Discontinuation of pembrolizumab, ventilatory support, and treatment with systemic and inhaled corticosteroids resulted in resolution of respiratory failure; however, her underlying asthma remains poorly controlled. Conclusion: To our knowledge, this case is the first report of pembrolizumab-induced obstructive respiratory failure in a patient being treated for RRP.

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Alpha-Interferon (Wellferon) as an Adjunct to Standard Surgical Therapy in the Management of Recurrent Respiratory Papillomatosis

Annals of Otology Rhinology & Laryngology ◽

10.1177/000348948809700409 ◽

1988 ◽

Vol 97 (4) ◽

pp. 376-380 ◽

Cited By ~ 14

Author(s):

Bruce N. Benjamin ◽

Henley Harrison ◽

Paul A. Gatenby ◽

Kaye Cameron ◽

Robert Kitchen ◽

...

Keyword(s):

Complete Remission ◽

Crossover Study ◽

Total Dose ◽

Laser Therapy ◽

Partial Remission ◽

Recurrent Respiratory Papillomatosis ◽

Alpha Interferon ◽

High Dose ◽

Daily Dose

Ten patients received lymphoblastoid alpha-interferon (Wellferon) in a crossover study so that Wellferon and standard microsurgical laryngeal laser therapy could be compared to laser therapy alone. Wellferon was administered initially at an intravenous high dose of 15 megaunits/m2 for 5 days followed by a daily dose of 2 megaunits/m2 subcutaneously for 6 months. Dosage was adjusted according to predefined toxicity. One patient was withdrawn from the study. Of the others, all but one received over 75 % of the planned total dose. At follow-up of the nine assessable patients, complete remission was achieved in two of them, partial remission in four, and no response in the remainder. The two complete remissions were sustained for 2 years, but the four partial remissions were not sustained. Thus, a role for alpha-interferon in the kind of regimen used here remains to be established.

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