Ann Thorac Surg 1995;60:1806-1807
© 1995 The Society of Thoracic Surgeons
Case Report
Surgical Management of Pulmonary Carcinoma Secondary to Recurrent Respiratory Papillomatosis
Andreas Sakopoulos, MD,
Kenneth A. Kesler, MD,
Edward C. Weisberger, MD,
Mark W. Turrentine, MD,
Dewey J. Conces, Jr, MD
Cardiothoracic Division, Department of Surgery, and Departments of Otolaryngology and Radiology, Indiana University School of Medicine, Indianapolis, Indiana
Accepted for publication June 6, 1995.
 |
Abstract
|
|---|
Recurrent respiratory papillomatosis is a rare, but acknowledged, risk factor for pulmonary squamous cell carcinoma. Although previous reports suggest a poor prognosis for lung cancer associated with papillomatosis, we have successfully treated 1 such patient, who presented with three synchronous pulmonary malignancies, using parenchyma-sparing resection techniques.
|
Introduction
|
|---|
The patient was an 18-year-old female nonsmoker who had been treated for recurrent respiratory papillomatosis (RRP) since 6 months of age. She had undergone more than 50 laryngoscopies for papilloma excision and was known to have pulmonary involvement by chest roentgenographs since the age of 14 years. Ultimately, left-sided pleuritic chest pain, intermittent hemoptysis, and fever with leukocytosis developed. Several weeks of antibiotic trials resulted in only partial symptomatic improvement. A chest computed tomographic scan demonstrated a large mass in the left lower lobe (Fig 1
). Note was made of a much smaller nodule, which had been present at the site of this mass on a computed tomographic scan obtained 1 year before this study. Two other soft tissue masses were present, which had developed at the sites of previous cysts, and demonstrated no change in size since the prior examination (Fig 2).
View larger version (111K):
[in this window]
[in a new window]
|
Fig 1. . Computed tomographic scan demonstrates a large soft tissue mass in the left lower lobe. A thoracotomy this was found to be a squamous cell carcinoma.
|
|
View larger version (118K):
[in this window]
[in a new window]
|
Fig 2. . Computed tomographic scan at the level of the inferior pulmonary veins. Carcinoma in the right lower lobe appears as an irregular opacity arising from the wall of a cyst (arrow). Also seen are a cyst containing fluid in the left lower lobe and a small papilloma in the right lower lobe (arrowhead).
|
|
In light of her refractory symptomatology and documented evidence of significant growth, she underwent left lower lobectomy with ipsilateral mediastinal lymph node dissection. Pathologic analysis revealed a grade II, moderately differentiated squamous cell carcinoma without nodal metastases. Within 2 weeks of an uneventful recovery she underwent percutaneous fine-needle aspiration biopsy of both right-sided lesions which were also positive for squamous cell carcinoma. A right upper lobe posterior segmentectomy and right lower lobectomy were performed, again with mediastinal lymph node dissection. The posterior segment was cleaved from the remainder of the right upper lobe with electrocautery, leaving a 1-cm tumor-free margin.
Her postoperative course was complicated by a parenchymal air leak, which resolved in 3 weeks. Pathologic analysis again revealed a moderately differentiated squamous carcinoma with intrabronchial papillomatosis. All lymph nodes and surgical margins were free of microscopic tumor. Final pathologic staging of all three lung neoplasms was T2 N0, or stage I. Preoperative forced expiratory volume in 1 second and forced vital capacity were 2.3 and 3.1 L, respectively, with little change 3 years after at 2.2 and 3.0 L. She has been followed up at routine intervals with chest radiographs and is disease free after 3 years. She currently attends college, actively participating in intramural sports, and has recently carried a normal pregnancy to term.
|
Comment
|
|---|
Recurrent respiratory papillomatosis is the most common pediatric benign respiratory tract neoplasm, with 1,500 new cases diagnosed each year [1, 2]. Human papilloma virus, particularly strains 6 and 11, has a trophism for squamous epithelial cells, inducing cellular proliferation or even frank dysplasia [3]. Macroscopically, multiple small villous proliferations develop, mainly in the upper airways, resulting in voice changes; however, occasionally proliferation may be severe enough to cause airway obstruction requiring tracheostomy. Infected patients typically require repeated surgical excisions to control symptoms. In approximately 5% of patients with RRP, lower airway involvement develops by viral dissemination, and less than 1% progress to pulmonary parenchymal involvement [5]. Pulmonary lesions radiographically appear as either solid or cystic masses. Occasionally large distal intrabronchial papillomas cause obstructive radiographic abnormalities including atelectasis, pneumonia, abscess, or bronchiectasis.
The development of laryngeal or lung cancer is not unusual in patients with RPR who have had tobacco or radiation exposure, the latter of which was historically used as a treatment modality [1, 6]. In contrast, however, there exist only 10 reported cases of RPR patients in whom carcinoma has developed without synergistic risk factors [1, 3, 5]. These patients characteristically manifested RRP before 2
years of age (mean age, 2.3 years) with progressive illness requiring frequent papilloma excisions and tracheostomy. Pulmonary squamous cell carcinoma developed one to two decades (mean, 18.7 years) after the onset of symptoms without apparent exposure to any promoting factors. All of these pulmonary neoplasms were reported to be of a moderate to well-differentiated histologic grade. Most of these patients, however, had ``advanced'' disease when the diagnosis of malignancy was established, and operation therefore was rarely offered.
Our patient with RRP underwent staged bilateral thoracotomies for surgical extirpation of multifocal pulmonary squamous cell carcinoma. The other reported survivor in this subset of RPR patients underwent an initial lobectomy, but was returned to operation 1 week later for chest wall excision after microscopy revealed this mass to be an unanticipated malignancy with surgical margin involvement [5]. This case, as well as the finding of carcinoma in the two radiographically stable right-sided lesions in our patient, underscores the importance of maintaining a high index of suspicion for malignant degeneration with any pulmonary involvement in these extremely young patients. Despite the grim reported outcome of many patients with RRP complicated by pulmonary squamous cell carcinoma, these 2 cases demonstrate that a favorable outcome can occur.
Patients with severe long-standing RRP should be radiographically monitored at regular intervals for pulmonary involvement and further evaluation actively pursued if any suspicion of malignancy arises. Fine-needle aspiration biopsy of solid parenchymal lesions should allow cytologic differentiation between benign papilloma and carcinoma. Aggressive surgical resection with parenchyma-sparing techniques of multiple lesions can result in at least medium-term freedom from disease, which is probably attributable to the low-grade nature of these neoplasms.
|
Footnotes
|
|---|
Address reprint requests to Dr Kesler, 545 Barnhill Dr, EM 212, Indianapolis, IN 46202.
|
References
|
|---|
- Schnadig VJ, Clark WD, Clegg TJ, Yao CS. Invasive papillomatosis and squamous carcinoma complicating juvenile laryngeal papillomatosis. Arch Otolaryngol Head Neck Surg 1986;112:966–71.[Abstract/Free Full Text]
- Kashima HK, Shah F, Shah K, et al. A comparison of risk factors in juvenile-onset and adult-onset recurrent respiratory papillomatosis. Laryngoscope 1992;102:9–13.[Medline]
- Byrne JC, Tsao MS, Fraser RS, Howley PM. Human papilloma virus-11 DNA in a patient with chronic laryngotracheobronchial papillomatosis and metastatic squamous-cell carcinoma of the lung. N Engl J Med 1987;31:873–8.
- Kramer SS, Wehunt WD, Stocker JT, et al. Pulmonary manifestations of juvenile laryngotracheal papillomatosis. AJR 1985;144:687–94.[Abstract/Free Full Text]
- Helmuth RA, Strate RW. Squamous carcinoma of the lung in a nonirratiated, nonsmoking patient with juvenile laryngotracheal papillomatosis. Am J Surg Pathol 1987;11:643–50.[Medline]
- Cripe TP. Human papillomaviruses: pediatric perspectives on a family of multifaceted tumorigenic pathogens. Pediatr Infect Dis J 1990;9:836–44.[Medline]
This article has been cited by other articles:
|
|
|
|
 
A. Gimenez, T. Franquet, R. Prats, P. Estrada, J. Villalba, and S. Bague
Unusual Primary Lung Tumors: A Radiologic-Pathologic Overview
RadioGraphics,
May 1, 2002;
22(3):
601 - 619.
[Abstract]
[Full Text]
[PDF]
|
|
|
Top
Abstract
Introduction
