Ann Thorac Surg 2008;86:1025-1026. doi:10.1016/j.athoracsur.2008.02.073
© 2008 The Society of Thoracic Surgeons
Case Reports
A Rare Case of Primary Pleomorphic Adenoma in Main Bronchus
Jason Fitchett, MBBCha,*,
Heyman Luckraz, FRCSa,
Allen Gibbs, FRCPathb,
Peter O'Keefe, FRCSa
a Cardiothoracic Unit, University Hospital of Wales, Heath Park, Cardiff, United Kingdom
b Histopathology Department, University Hospital of Wales, Heath Park, Cardiff, United Kingdom
Accepted for publication February 25, 2008.
* Address correspondence to Dr Fitchett, Cardiothoracic Unit, Block C5, University Hospital of Wales, Heath Park, Cardiff, CF14 4XN, United Kingdom (Email: jasonfitchett{at}hotmail.com).
| Mr O'Keefe discloses that he has a financial relationship with Edwards Lifesciences and Baxter Healthcare.
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Abstract
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Primary pleomorphic adenoma of the lung is a rare occurrence. Clinical suspicion is normally low due to its rarity. We describe a case of a primary pleomorphic adenoma arising from the origin of the right main bronchus and include our management strategy.
Pleomorphic adenoma, although a relatively common occurrence in the head and neck, is a rarity in the lungs [1–4]. The typical appearance is of proliferation of spindle-shaped cells arranged in glandular sheet-like structure embedded in myxomatous stroma [4] (Figs 1, 2).
Due to the low incidence in the lungs there is a lack of consensus with regard to management, prognosis, and survival. Therefore, it is important to highlight these cases to increase knowledge. The majority of pleomorphic adenomas arising in the lungs are benign and slow growing [2]. They are believed to have arisen from bronchial gland epithelium [1, 5].
A 65-year-old man was referred with a 5-month history of a hoarse barking cough. He was a lifelong nonsmoker. Ear, nose, and throat examination had revealed no oropharyngeal cause. However, he did have mild audible stridor and distended veins on the right anterior thorax with widespread referred rhonchi on the right, but no lymphadenopathy or weight loss. Chest x-ray films were normal. A computed tomographic scan showed a 13-mm intraluminal mass in the right main bronchus (Fig 3). The differential diagnosis at this stage included both benign and malignant lesions, including carcinoids and pleomorphic adenoma. Flexible bronchoscopy revealed a lesion at the origin of the right main bronchus, which was highly suspicious of adenocarcinoma (Fig 4). However, due to the risk of hemorrhage during biopsy, the patient was referred for rigid bronchoscopy and biopsy. Rigid bronchoscopy revealed a fleshy polypoid mass arising from the carinal output on the right main bronchus occluding the right main bronchus. The mass was easily removed with a diathermy snare without significant bleeding. No other tracheobronchial lesions or extrinsic compressions were found. The histologic diagnosis confirmed a primary pleomorphic adenoma, a very rare finding in this location.
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Fig 3. Computed tomographic thoracic image showing pedunculated intraluminal lesion at the origin of the right main bronchus.
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Comment
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Pleomorphic adenoma, also known as mixed tumor, although common in the head and neck in the salivary glands is a very rare occurrence in the lung [1–4]. There are very few case reports in the literature of pulmonary pleomorphic adenoma, and no definitive incidence has been recorded. From the few care reports in the literature, pleomorphic adenomas of the lung typically affect people between the ages of 35 to 74. However, a case has been described in an 8-year-old boy [2]. It is believed to have contributed to 1% of lung carcinomas and between 2% to 9% of all pleomorphic adenomas [5]. Approximately one third of cases are found incidentally [3]. The majority of patients do presents with symptoms. The most common symptoms are cough, sputum production, dyspnea, wheeze, and stridor [1, 2, 3, 5]. Due to the lack of information, it is difficult to assess prognosis when a pleomorphic adenoma arises in the lung [2, 3]. The most reliable prognostic features are size, extent of local infiltration, and degree of mitotic activity [5]. However, the recurrence rate is notoriously high for pleomorphic adenomas of the salivary glands [3]. Radiotherapy is believed to have only offered temporary palliation [5].
In conclusion, pleomorphic adenoma of the lung, particularly a primary lesion, is a very rare occurrence. However, it does have a common group of presenting symptoms arising from the obstruction of the bronchus caused by the intraluminal mass. Although rare, it is worth considering pleomorphic adenoma when presented with a pedunculated intraluminal lesion. It is only by monitoring that we can truly appreciate the incidence, prognosis, and optimum management of this rare occurrence.
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References
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- Moran CA, Suster S, Askin FB, Koss MN. Benign and malignant salivary gland-type mixed tumors of the lung Cancer 1994;73:2481-2490.[Medline]
- Baghai-Wadji M, Sianati M, Nikpour H, Koochekpour S. Pleomorphic adenoma of the trachea in an 8-year-old boy: a case report J Pediatr Surg 2006;41:E24-E26.
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- Takeuchi E, Shimizu E, Sano N, Yamaguchi T, Yanagawa H, Sone S. A case of pleomorphic adenoma of the lung with multiple distant metastases Anticancer Res 1998;18:2015-2020.[Medline]
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Abstract
Introduction
