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Ann Thorac Surg 2000;70:292-295
© 2000 The Society of Thoracic Surgeons

Case report

Tracheobronchial glomus tumor

Thomas H. Lange, MDa, Mitchell J. Magee, MDa, Theresa M. Boley, MSNa, Stephen W. Bell, MDa, Stephen R. Hazelrigg, MDa

a Division of Cardiothoracic Surgery, Southern Illinois University School of Medicine, Springfield, Illinois, USA

Address reprint requests to Ms Boley, Division of Cardiothoracic Surgery, SIU School of Medicine, 800 N Rutledge, Room D314, PO Box 19638, Springfield, IL 62794-9638
e-mail: tboley{at}siumed.edu


   Abstract
Top
 Abstract
Introduction
 Comment
 References
 
Glomus tumors are uncommon. A review of the literature for tracheobronchial glomus tumors revealed 13 tracheal glomus tumors. The diagnosis may be elusive and so the true incidence of tracheobronchial glomus tumors may be greater than that reported. Three of the 14 glomus tumors were initially believed to be carcinoid. Glomus tumors should be included in the differential diagnosis of tracheobronchial tumors.


   Introduction
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 Abstract
 Introduction
Comment
 References
 
Glomus tumors are uncommon with an estimated incidence of 1.6% in 500 consecutive soft tissue tumors [1]. These tumors arise from cells that resemble the modified smooth muscle cells of the glomus body. The normal glomus body is a form of arteriovenous anastomosis used in thermal regulation. The most common location for glomus tumors is the subungual region of the finger, along with the palm, wrist, forearm, and foot [2]. They have also been reported in more unusual locations, including bone [3], stomach [4], rectum, mesentery [1], mediastinum [5], and trachea [6]. A total of 14 cases of glomus tumor located in the trachea have been previously reported. The known experience with glomus tumors of the tracheobronchial tree is reviewed.

The patient was a 20-year-old male who presented to the emergency room with a right upper quadrant abdominal gunshot wound. His past medical history was significant for a recent diagnosis of asthma, for which a single bronchodilator had been prescribed. During initial evaluation, a right tube thoracostomy was performed for hemothorax. He was taken to the operating room for an exploratory laparotomy with the findings of a small laceration to his right kidney and left lobe of the liver. On postoperative day number 2, the patient experienced an episode of severe dyspnea and was noted on chest x-ray to have significant left lung atelectasis. Bronchoscopy demonstrated a tan-yellow polypoid mass located in the proximal left bronchus. The lesion occupied approximately 80% of the lumen and created significant postobstructive atelectasis. Biopsy was performed with the initial diagnosis of typical carcinoid tumor. A computed tomography scan of the chest was obtained which demonstrated a polypoid lesion on the posterior aspect of the left bronchus just distal to the carina. No enlarged lymph nodes were noted. The patient was discharged home on hospital day number 10.

Following an uneventful recovery from the gunshot wound and abdominal surgery, the patient underwent a left bronchial sleeve resection, performed through a left anterior thoracotomy. The patient had an uneventful postoperative course and was discharged home on postoperative day number 4. He was seen in routine outpatient follow-up at 8 weeks following the resection. At that time, he had no respiratory complaints and had an unremarkable chest radiograph. Nine months postoperatively, he was again admitted as a trauma patient. At that time he had no respiratory complaints and a computed tomography scan of his chest revealed no suspicious masses or enlarged lymph nodes.

The surgical specimen of the bronchial sleeve resection, consisted of a cylinder of bronchus measuring 1.8 cm in length and 2.3 cm in circumference. Projecting into the bronchial lumen was a polypoid tan mass measuring 1.5 x 1.0 x 0.4 cm (Fig 1). The proximal and distal resection margins were free of tumor. Histologically, the tumor was composed of solid sheets of cells interrupted by a delicate vasculature. The tumor cells exhibited a rounded nucleus with faintly eosinophilic cytoplasm. Occasional large hyperchromatic nuclei, probably representing degenerative change, were present in some areas. Focally increased mitotic activity was also observed. Initial pathology results from permanent sections were consistent with the diagnosis of atypical carcinoid tumor. Further immunohistochemical staining was performed on both sleeve resection and bronchoscopy specimens leading to the final diagnosis of glomus tumor. The tumor cells exhibited strong reactivity for the muscle markers actin and desmin. Stains for neuroendocrine markers and cytokeratin were negative.



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  		Fig 1. Sagittal view of tumor projecting into the bronchial lumen.

 

   Comment
Top
 Abstract
 Introduction
 Comment
References
 
Glomus tumors are relatively uncommon soft tissue tumors exhibiting no gender predilection. The neoplastic cells are regarded as variants of smooth muscle cells. Unusual locations for glomus tumors have been reported as described above. A review of the literature demonstrated 13 glomus tumors of the trachea reported since Hussarek and Rieder’s report in 1950 (Table 1). Three females and ten males between the ages of 20 and 74 (median age 57) have been reported. Presenting symptoms include dyspnea, cough, hemoptysis, and stridor. Most lesions appear to be benign and have been adequately treated with local resection. A total of 4 patients, including ours, showed tumor involvement beyond the tracheal wall [7–9]. This finding suggests that endoscopic ablation may not be adequate treatment for these lesions and complete resection is recommended.


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  		Table 1. Glomus Tumors of the Respiratory Tract

 
The diagnosis of tracheobronchial glomus tumors can be elusive and, in fact, the incidence may be greater than reported due to incomplete characterization. Two of the reported 13 tracheal glomus tumors, and this bronchial tumor were initially diagnosed as carcinoid tumors [7, 10]. The bronchoscopic appearance is also suggestive of the more common carcinoid tumor. It was only after immunohistochemical staining or electron microscopy that the final diagnosis of glomus tumor was obtained. The routine histochemistry more closely resembles atypical carcinoid, which carries significantly different implications for treatment and prognosis.

The surgical treatment for atypical carcinoid tumors includes a more extensive resection of surrounding tissue along with mediastinal lymph node dissection [11–13]. In addition to wide resection, stage III and IV atypical carcinoid tumors should be treated with postoperative chemotherapy [11].

The surgical treatment for glomus tumors should be a limited resection with negative margins and no formal mediastinal lymph node dissection need be included. In the 14 cases of glomus tumors reviewed, there were no recurrences after local resection. There is no evidence supporting a role for chemotherapy in the treatment of tracheobronchial glomus tumors. Glomus tumors should be included in the differential diagnosis of tracheobronchial lesions.


   Acknowledgments
 
The authors express their appreciation to Patricia Schafer for her assistance in the preparation of this manuscript.


   References
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 Abstract
 Introduction
 Comment
 References
 

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  3. Smyth M. Glomus-cell tumors in the lower extremity. J Bone Joint Surg Am 1971;53:157-159.[Free Full Text]
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  8. Garcia-Prats M.D., Sotelo-Rodriquez M.T., Ballestin C., et al. Glomus tumour of the trachea. Histopathology 1991;19:459-464.[Medline]
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Accepted for publication December 1, 1999.




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