Tracheal Papillomatosis with Multiple HPV Serotypes

Labib Gilles Debiane, MD; Lorenzo Zaffiri, MD; Casey Stahlheber, MD

INTRODUCTION: Adult-onset recurrent respiratory papillomatosis (AORRP) is a rare cause of upper airway obstruction. We describe a unique case of near-total tracheal obstruction from multi-serotype tracheal papillomatosis presenting as poorly controlled asthma and hemoptysis.

CASE PRESENTATION: A 59-year-old Hispanic man presented with a 10-day history of hemoptysis. He had been recently diagnosed with asthma due to non-resolving cough and dyspnea. Computed tomography of the chest revealed a large tracheal mass. Bronchoscopy showed a large polypoid tumor arising circumferentially from the tracheal wall with near-total obliteration of the lumen. The tumor was debulked in 2 stages using argon plasma coagulation (APC) and cryotherapy, reducing the narrowing to <30% initially then <10% subsequently. Interestingly, histopathology of the tracheal mass showed squamous papilloma positive for Human Papilloma Virus (HPV) 6, 11, 16, and 18 serotypes. On follow up, asthma symptoms had resolved.

DISCUSSION: AORRP is a rare but usually benign condition. It presents with a wide variety of symptoms like cough, dyspnea, stridor, and hemoptysis. It is caused by HPV, with serotypes 6 and 11 accounting for the majority of cases. These serotypes have low risk for malignant transformation. HPV 16 and 18 are associated with severe dysplasia and malignancy. Simultaneous co-infection with 4 HPV serotypes has not been described. To date, there is no curative treatment mostly due to persistence of HPV in the adjacent, apparently normal, mucosa. Combined approaches were studied with limited success, including recurrent surgical interventions and treatment with immunomodulators, antiviral, and chemotherapy drugs. Growing literature supports using APC and cryotherapy to debulk endobronchial lesions thanks to a good safety profile, effective hemostasis, and widespread availability. The quadrivalent HPV vaccine is shown to prevent genital tract HPV-related infection and neoplasia. During a 1-year follow up, a recent prospective study found no recurrence of the respiratory papillomatosis in 85% of vaccinated patients. On the basis of these studies and this unique co-infection with multiple serotypes, we opted to administer the quadrivalent vaccine.

CONCLUSIONS: Tracheal papillomatosis can mimic common pulmonary diseases and should be considered when diseases do not respond to usual therapies. Co-infection with multiple serotypes is uncommon.

Chest. 2014;146(4_Meeting