Recurrent hemoptysis in a 62-year-old smoker.

Tracheal papillary adenoma is a rare benign tumor. We report a case of papillary adenoma in a 62-year-old male smoker who presented with recurrent hemoptysis. The tumor was located in the upper third of trachea and forceps biopsy through flexible bronchoscopy was uncomplicated and diagnostic.

INTRODUCTION

Of all the primary respiratory tract tumors, only 2% occur in trachea.[1] In adults, 80–90% of primary tracheal tumors are malignant.[2] The most common benign tracheal tumor is papilloma followed by fibroma and haemangioma; others like lipoma, adenoma, hamartoma, leiomyoma are very rare.[3] Very few cases of papillary adenoma have been reported.[45] Surprisingly, even large studies of tracheal tumors failed to document even a single case of tracheal papillary adenoma.[67] We report a case of benign tracheal papillary adenoma in an adult as rare occurrence.

CASE REPORT

A 62-year-old male presented with recurrent hemoptysis for the past 1 year. Hemoptysis was intermittent, minimal and controlled with conservative treatment. There was no significant cough, breathlessness or wheezing. Medical history was not significant. He worked as a fireman and smoked 20–25 cigarettes per day over the last 40 years. Clinical examination was unremarkable. Being a chronic heavy smoker, pulmonary carcinoma was initially suspected and patient was admitted. Radiograph and computed tomography (CT) of the chest revealed no abnormality. Cardiac evaluation was negative. At flexible bronchoscopy, a reddish, round and smooth tumor mass was seen in the right lateral wall of the upper third of the trachea [Figure 1]. Multiple biopsies of the lesion were obtained without significant bleeding from biopsy site. Ultrasound neck showed a mass lesion (1.91 cm × 1.9 cm) adjacent to the trachea involving its wall and extending into its lumen on the right side [Figure 2]. A contrast enhanced computed tomography (CECT) showed moderate enhancement of the mass lesion [Figure 3]. Histological examination of bronchial biopsy confirmed the mass as papillary adenoma [Figures 4 and 5]. Patient was referred to cardiothoracic surgeon for further management. However, the patient decided to undergo surgery only when hemoptysis recurs and remained well during the follow-up period of 9 months.

Figure 1

Endotracheal lesion as seen through flexible bronchoscope

Figure 2

Ultrasound of the neck showing mass lesion adjacent to the lateral and posterior portion of trachea extending into its lumen

Figure 3

Axial contrast enhanced computed tomography of neck section showing moderate enhancement of lesion

Figure 4

Smear showing tumor cells arranged in glandular and papillary pattern (H and E, ×100)

Figure 5

Smear showing vacuolated cytoplasm and basally pushed nuclei (H and E, ×400)

DISCUSSION

Primary benign tracheal tumors can arise from the respiratory epithelium, salivary glands and mesenchymal structures of the trachea. The benign tumors originating from the surface epithelium include squamous papilloma, transitional cell papilloma and papillary adenoma.[8] In our case, papillary adenoma was diagnosed on the basis of smear section showing tumor cells arranged mostly in glandular pattern with occasional foci of tumor cells arranged in papillary pattern along with fibrovascular core. Some of the tumor cells lining the glands had vacuolated cytoplasm with basally placed nuclei. There was no evidence of mitotic activity. These features differentiated papillary adenoma from papilloma, hamartoma, pleomorphic adenoma, papillary carcinoma of thyroid. Compared to malignant tracheal tumors which cause symptoms earlier due to their rapid growth, benign tracheal tumors cause symptoms late resulting in delayed diagnosis by months to years.[9] A benign tracheal tumor may not cause any symptom until at least 70% of tracheal lumen is occluded.[10] The symptoms are non-specific and include dyspnoea, cough and hemoptysis. In our case, recurrent minimal hemoptysis was the only symptom, which seems to result irrespective of the size of tracheal tumor. Diagnosis of benign tracheal tumors are normally done through flexible bronchoscopy and rigid bronchoscopy is usually done if additional procedures like electrocautery are planned.[11] In our case, the tumor had grown through the wall of trachea and larger portion is located adjacent to the trachea. CT features in benign tracheal tumors are non-specific. Sometimes, the typical CT appearances may suggest the diagnosis. For example, a fat containing tumor of heterogenous soft-tissue density with islands of fatty tissue is suggestive of fibrolipoma or with areas of fat density and calcified foci as in hamartoma.[12] In our case, the tumor did not have fat attenuation and moderate enhancement on CECT study, raised the suspicion of it being malignant. However, the histological findings were typical of a papillary adenoma. Multi detector CT is considered as imaging technique of choice for detecting and staging of central airway tumors and it can accurately determine the intraluminal and extraluminal extension of tumor as well as post obstructive complications such as atelectasis, pneumonia.[13] A tracheal tumor may grow into or through the tracheal wall making complete surgical resection difficult.[14] Benign primary tracheal tumors are best treated by surgical resection with reconstruction of airway; other treatment options being cryotherapy, electrocoagulation and laser treatment through bronchoscopy.[15]