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A Unique Case of Chronic Cough

      A 49-year-old man with a history of coronary artery disease presented to pulmonary clinic with a progressive chronic cough that started 1 year previously, associated with scant hemoptysis and recent onset dyspnea on exertion. He denied smoking, fevers, night sweats, and weight loss. Results of the physical examination were unremarkable. Chest computed tomography (Figure 1) showed a large longitudinal filling tracheal defect. Follow-up bronchoscopy revealed several large wart-like nodular lesions on the vocal cords and in the trachea (Figure 2), causing severe tracheal narrowing. The lesions were removed with electrocautery and snare to ensure near complete patency of trachea. Pathology results of the tracheal lesions returned with squamous papilloma positive for human papilloma virus (HPV)-6 and 11, consistent with a diagnosis of diffuse laryngotracheal papillomatosis (LTP).
      Figure thumbnail gr1
      Figure 1Longitudinal filling defect in the trachea (black arrow) seen prominently on coronal images of the chest computed tomography.
      Figure thumbnail gr2
      Figure 2Bronchoscopic visualization of large, nodular papillomas on the vocal cords (A) and throughout the trachea (B), causing significant obstruction.
      Diffuse LTP is the benign, neoplastic, papillomatous epithelial growth caused by HPV infection, most commonly HPV-6 and 11. Symptoms depend on the severity of the papillomas and include cough, dyspnea, voice change, and—rarely—stridor or upper-airway obstruction.
      • Harris K.
      • Chalhoub M.
      Tracheal papillomatosis: what do we know so far?.
      • Ribeiro Fortes H.
      • von Ranke F.M.
      • Escuissato D.L.
      • et al.
      Recurrent respiratory papillomatosis: a state-of-the-art review.
      • Kalanjeri S.
      • Hoffman S.
      • Farver C.
      • Almeida F.A.
      Diffuse tracheal papillomatosis.
      The diagnosis is made via direct visualization and biopsy.
      • Harris K.
      • Chalhoub M.
      Tracheal papillomatosis: what do we know so far?.
      ,
      • Ribeiro Fortes H.
      • von Ranke F.M.
      • Escuissato D.L.
      • et al.
      Recurrent respiratory papillomatosis: a state-of-the-art review.
      Unfortunately, the disease has a chronic course of recurrent papillomas.
      The primary treatment is endobronchial debulking and debridement via bronchoscopy, including microdebrider, electrocautery, argon plasma coagulation, and cryotherapy. The use of laser therapy has diminished owing to the concern for HPV transmission via the vapor plume.
      • Ribeiro Fortes H.
      • von Ranke F.M.
      • Escuissato D.L.
      • et al.
      Recurrent respiratory papillomatosis: a state-of-the-art review.
      ,
      • Kalanjeri S.
      • Hoffman S.
      • Farver C.
      • Almeida F.A.
      Diffuse tracheal papillomatosis.
      Recent advances in medical therapies for tracheal papillomatosis include HPV vaccination, cidofovir, and bevacizumab, with studies showing decreased tumor burden and increased intervals between therapeutic procedures after treatment
      • Mohr M.
      • Schliemann C.
      • Biermann C.
      • et al.
      Rapid response to systemic bevacizumab therapy in recurrent respiratory papillomatosis.
      ,
      • Hočevar-Boltežar I.
      • Matičič M.
      • Sereg-Bahar M.
      • et al.
      Human papilloma virus vaccination in patients with an aggressive course of recurrent respiratory papillomatosis.
      (Supplemental Figures 1 and 2, available online at http://www.mayoclinicproceedings.org).

      Supplemental Online Material

      Figure thumbnail figs1
      Supplemental Figure 1Tracheal defect additionally shown on both axial (A, black arrow) and sagittal (B, black arrow) view of the chest computed tomography.
      Figure thumbnail figs2
      Supplemental Figure 2Closer bronchoscopic imaging of the tracheal masses exemplify the wart-like, papillomatous appearance.

      References

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        Recurrent respiratory papillomatosis: a state-of-the-art review.
        Respir Med. 2017; 126: 116-121
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        Rapid response to systemic bevacizumab therapy in recurrent respiratory papillomatosis.
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        Human papilloma virus vaccination in patients with an aggressive course of recurrent respiratory papillomatosis.
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