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Ann Thorac Surg 2004;77:2201-2202
© 2004 The Society of Thoracic Surgeons

Case report

Human papillomavirus and squamous cell carcinoma in a solitary tracheal papilloma

Chung-Wah Lam, MDa, Alan Ronald Talbot, MDb,d, Kun-Tu Yeh, MDc, Sing-Chun Lina, Chia-En Hsieha, Hsin-Yuan Fang, MDa*

a Departments of Surgery, Changhua Christian Hospital, Changhua, Taiwan
b Critical Care Medicine, Changhua Christian Hospital, Changhua, Taiwan
c Pathology, Changhua Christian Hospital, Changhua, Taiwan
d Department of Internal Medicine, Chung Shan Medical University, Taichung, Taiwan

Accepted for publication June 13, 2003.

* Address reprint requests to Dr Fang, Department of Surgery, Changhua Christian Hospital, No. 135 Nanhsiao St, Changhua, 500 Taiwan
e-mail: 51302{at}cch.org.tw


   Abstract
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 Abstract
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We report the case of a 52-year-old woman suffering from breathlessness on exertion. A chest radiograph identified a lesion in the intrathoracic trachea. A tumor 0.4 x 0.5 x 0.7 cm in size causing an approximately 80% reduction in the cross-sectional area of the trachea 3 cm above the carina was removed, and histology showed moderately differentiated squamous cell carcinoma. Intrathoracic resection of the tumor and anastomosis was performed through a right lateral thoracotomy. The final histology examination showed atypical papilloma. Polymerase chain reaction–restriction fragment length polymorphism confirmed human papillomavirus-6b in the tumor. The patient remained well 18 months after surgical intervention without recurrence.


   Introduction
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 Abstract
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Papillomatosis presents as multiple lesions, but it is not common in the trachea; one estimate of its incidence in adolescents older than 15 years is 1.8 cases per 100,000 people [1]. Malignant transformation may be idiopathic or due to carcinogen exposure, immunosuppressants, radiation, or smoking [2, 3].

Tracheobronchial papillomas may be caused by human papillomaviruses (HPVs), either HPV-6 or HPV-11 [2, 3], and malignant degeneration into squamous cell carcinoma occurs in 3% to 5% of patients [1]. Solitary papilloma rather than multiple lesions of papillomatosis is extremely rare.

We present the case of an adult patient who had a solitary tumor obstructing the airway in the intrathoracic trachea. The initial diagnosis was squamous cell carcinoma, but histologically, the base of the tumor was atypical papilloma.

A 52-year-old woman complained of breathlessness on exertion, which had worsened over the previous 2 years. Her local physician had treated her with steroids for asthma, until an intratracheal lesion was identified on a chest radiograph.

The patient was referred to our clinic. She was a nonsmoker and did not work in any occupation associated with carcinogenic agents. Stridor was evident.

Flexible bronchoscopy (Fig 1A) and chest computed tomography (CT) (Fig 1B) documented a cauliflower-like, warty tumor. The tumor measured 0.4 x 0.5 x 0.7 cm and protruded into the trachea, resulting in an approximately 80% reduction of the cross-sectional area 3 cm above the carina. Pulmonary function studies showed a flow volume loop pattern consistent with intrathoracic upper airway obstruction.



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  		Fig 1. Flexible bronchoscope (A) and chest computed tomography (B) show a tumor 0.4 x 0.5 x 0.7 cm in size, 3 cm proximal to the carina.

 
The tumor was removed through a rigid bronchoscope under general anesthesia. Histologically, the specimen showed moderately differentiated squamous cell carcinoma consisting of large, keratinized epidermoid cells with papillary growth pattern transformation (Fig 2A).



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  		Fig 2. (A) Moderately differentiated squamous cell carcinoma consisting of large, keratinized epidermoid cells with papillary growth pattern (hematoxylin & eosin stain, magnification x200). (B) Atypical squamous cell papilloma is covered by atypical squamous epithelium (hematoxylin & eosin stain, magnification x100). (C) The results of polymerase chain reaction–restriction fragment length polymorphism from the biopsy specimen are positive for human papillomavirus-6b. (BamHI = Bacillus amyloliquefaciens; Ddel = Desulfovibrio desulfuricans; Dral = Deinococcus radiophilus; EcoRI = Escherichia coli; HaeIII = Haemophilus aegyptius; M = maker; Pst1 = Proviencia stuartii; UC = control.)

 
Because of cushingoid changes from the steroids, a 2-month time interval was used to wean the patient off steroids before proceeding to a second operation. In this operation, the intrathoracic resection of the tracheal tumor was accomplished by the removal of three cartilage rings, and after frozen section confirmed that the margins were free of atypical cells, anastomosis was performed through a right lateral thoracotomy.

Histologically, the tumor consisted of atypical squamous cell papilloma composed of loose fibrovascular cores covered by atypical squamous epithelium (Fig 2B). Polymerase chain reaction–restriction fragment length polymorphism (PCR-RFLP) confirmed the presence of HPV-6b in the tumor specimens (Fig 2C).

Postoperatively, the patient gained immediate relief from her symptoms. She remained well 18 months after the operation, with outpatient follow-up with regular bronchoscopic examination continuing to show that she remains free of recurrence.


   Comment
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HPV causes respiratory tract papillomatosis (multiple lesions) or papilloma (single lesion). Onset during adulthood is more common among men and often occurs in the third or fourth decade of life [1, 4].

Dyspnea on exertion and hoarseness are the most common symptoms, often accompanied by stridor or wheezing. Other less common symptoms are a change of the voice, a chronic cough, hemoptysis, repeated respiratory infection, choking, and a feeling of something obstructing the throat [1, 4].

A chest radiograph may show a tracheobronchial lesion, atelectasis (segmental or lobar), or obstructive pneumonia. CT is a good choice for upper airway lesions, documenting the size, location, and involvement of surrounding structures. When upper intrathoracic airway obstruction occurs, pulmonary function studies reflect its severity, with flattening in inspiratory or/and expiratory phases.

Surgical resection is the preferred therapy for primary airway tumors such as squamous cell carcinoma and papilloma [1, 4]. Other treatment options include repeated laser therapy and photodynamic therapy with photosensitizing agents such as dihematoporphyrin ether, intralesional cidofovir, systemic and intralesional interferon, indole 3-carbinol, cimetidine, acyclovir, and retinoic acid [1].

Histology shows papillary lesions with keratinized epidermoid cells. The degrees of metaplasia and dysplasia are an index of malignant transformation. The viral infected pattern, koilocytes, is also found. The typing of the virus by PCR-RFLP or other molecular biologic methods may have a role in determining a prognosis [2, 3, 5]. Liu [6] studied 16 patients with recurrent respiratory papillomatosis using a slot blot hybridization technique and found HPV-6b in 87.5% of them, HPV-11 in 93.7%, and HPV-16 in 81.6%. The PCR-RFLP method confirmed the presence of HPV-6b in the tumor of our patient, with transformation into squamous cell carcinoma.

The malignant transformation of upper respiratory tract papillomatosis to squamous cell carcinoma is rare and most often occurs in patients with histories of smoking or radiotherapy [7]. The absence of risk factors for malignant transformation in some cases (including our patient) suggests that malignant transformation may occur spontaneously [3].

Our case was rare because the lesion was a single tumor, whereas papillomatosis usually occurs as multiple lesions; the patient was female, whereas HPV is more common among men; the patient was older than the usual adult age group for this disease; and the HPV was associated with malignant degeneration into squamous cell carcinoma.

The patient had an excellent outcome after the resection of the affected portion of the trachea. There was no evidence of recurrence after 18 months of follow-up, but we consider continued follow-up necessary.


   References
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 Abstract
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 Comment
 References
 

  1. Eloise M. Recurrent respiratory papillomatosis. eMedicine 2003. Available at: http://www.emedicine.com/med/topic2535.htm
  2. Byrne J.C., Tsao M.S., Fraser R.S., Howley P.M. Human papillomavirus-11 DNA in a patient with chronic laryngotracheobronchial papillomatosis and metastatic squamous-cell carcinoma of the lung. N Engl J Med 1987;317(14):873-878.[Medline]
  3. Rady P.L., Schnadig V.J., Weiss R.L., et al. Malignant transformation of recurrent respiratory papillomatosis associated with integrated human papillomavirus type 11 DNA and mutation of p53. Laryngoscope 1998;108(5):735-740.[Medline]
  4. Wood D.E. Management of malignant tracheobronchial obstruction. Surg Clin North Am 2002;82(3):621-642.[Medline]
  5. Harada H., Miura K., Tsutsui Y., et al. Solitary squamous cell papilloma of the lung in a 40-year-old woman with recurrent laryngeal papillomatosis. Pathol Int 2000;50(5):431-439.[Medline]
  6. Liu D.J. Human papillomavirus-DNA in laryngeal papilloma detected by nucleic acid hybridization, and the clinical treatment. Zhonghua Er Bi Yan Hou Ke Za Zhi 1993;28(6):358-359.[Medline]
  7. Guillou L., Sahli R., Chaubert P., et al. Squamous cell carcinoma of the lung in a nonsmoking, nonirradiated patient with juvenile laryngotracheal papillomatosis. Evidence of human papillomavirus-11 DNA in both carcinoma and papillomas. Am J Surg Pathol 1991;15(9):891-898.[Medline]



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