Ann Thorac Surg 2000;70:295-297
© 2000 The Society of Thoracic Surgeons
Case report
Glomus tumor of the trachea
Yasser M. Menaissy, MDa,b,c,
Anthony A. Gal, MDa,b,c,
Kamal A. Mansour, MDa,b,c
a Joseph B. Whitehead Department of Surgery, Atlanta, Georgia, USA
b Department of Pathology, Emory University School of Medicine, Atlanta, Georgia, USA
c Department of Laboratory Medicine, Emory University School of Medicine, Atlanta, Georgia, USA
Address reprint requests to Dr Mansour, Department of Surgery, The Emory Clinic, 1365 Clifton Rd NE, Atlanta, GA 30322
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Abstract
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Extracutaneous glomus tumors are unusual and their occurrence in the trachea has been recognized with extreme rarity. We present a case of surgically resected glomus tumor of the trachea in a 34-year-old man who presented with hemoptysis and who was initially diagnosed as having a carcinoid tumor.
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Introduction
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Only nine glomus tumors of the trachea have been reported among large series of tracheal resection and in case reports [1–9]. The clinical and histopathologic features of this rare benign tumor have been presented but are not well known [2, 8]. We present the case of a young man who underwent tracheal resection for a primary glomus tumor with clinical and histopathologic verification.
The patient, a 34-year-old Egyptian, nonsmoker, first presented to his primary care physician 2 months before operation with recurrent attacks of hemoptysis. He did not complain of dyspnea or chest pain. Preoperative chest radiographs and computed tomographic series revealed a mass lesion in the posterior wall of the trachea, extending posteriorly outside the trachea with possible esophageal involvement (Fig 1). The mass was 6 cm below the vocal cords, 4.5 cm above the carina, and extended 2.5 cm distally. Barium swallow showed smooth indentation without invasion of the cervical esophagus (Fig 2). Bronchoscopy showed an irregular hemorrhagic mass located in the posterior wall of the trachea at a distance of 6 cm below the vocal cords, and biopsy specimens initially diagnosed the mass to be a carcinoid tumor of the trachea. The patient was then referred to our institution for definitive treatment.
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Fig 1. Computed tomography scan of the trachea shows a mass lesion in the posterior wall of the trachea, causing 50% occlusion of lumen and extending to vertebral column.
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On August 26, 1999, bronchoscopy was performed to confirm the location of the lesion. Following this, general anesthesia was induced with sodium thiopental and fentanyl. An oral tracheal tube was placed after muscle paralysis with rocuronium. A tracheal tube was placed proximal to the tumor with the aid of a fiber-optic bronchoscope. A median sternotomy and transverse cervical incision was made, as the tumor was located at the thoracic inlet. Tracheal resection was performed, encompassing the tumor-bearing area and consisting of five tracheal rings. Frozen section analysis confirmed negative margins. The trachea was primarily anastomosed using interrupted 3-0 Vicryl (Ethicon, Somerville, NJ) sutures and then a pericardial flap was elevated and interposed between the innominate artery and the suture line. At the end of the procedure, fiberoptic bronchoscopy was performed to check the suture line and confirm its integrity. The patient was then awakened, extubated and two sutures of No. 1 Prolene (Ethicon, Somerville, NJ) were inserted between the chin and chest to keep the neck in a flexed position. Postoperatively, the patient did very well and a tomogram before his hospital discharge on the seventh postoperative day showed a good tracheal air column.
The gross tracheal specimen measured 3.5 cm in length with a luminal diameter of 2.0 cm. An exophytic 2.4 x 2.1 cm pink-tan mass extended through the tracheal wall into the posterior soft tissues, but did not involve the surgical resection margin. Microscopic analysis revealed that the tumor cells, arranged in nests or sheets, were surrounded by thin- and thick-walled blood vessels and not by thin-wall capillaries (Fig 3). The bland cells exhibited oval-to-round nuclei, and absence of hemorrhage, necrosis, mitoses, or pleomorphism (Fig 3 inset). The tumor cells were positive for smooth muscle actin and type IV collagen; the latter delineating strong staining at the interface between the cells. Immunostains for chromogranin and cytokeratin (AE 1-3) were negative.
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Fig 3. Representative low power magnification of tracheal glomus tumor demonstrating the tumor cells surrounded by prominent thick-walled blood vessels (hematoxylin and eosin, x40). (Inset) Representative high power magnification of tracheal glomus tumor demonstrating a solid nested pattern of bland tumor cells (hematoxylin and eosin, x40).
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Comment
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Glomus tumors are uncommon, benign neoplasms which are commonly seen in the extremities, particularly in the subungual region of the finger. This tumor was first described by Masson in 1924 [10]. Since then, sporadic reports of glomus tumors of varying types and locations have appeared in the literature. The tumor is a distinctive type of vascular tumor whose cell type is a modified smooth muscle cell closely resembling the glomus body from which the name is derived. These tumors share several morphologic features with carcinoid tumors. According to the relative proportions of glomus cells, vascular structures, and smooth muscle tissue, glomus tumors have been divided into three subtypes: classic glomus tumors, glomangiomas, and glomangiomyomas. The trachea is a very unusual and rare site for this tumor and only 9 cases of tracheal glomus tumors have been reported in the literature (Table 1).
The prominent nested pattern of bland tumor cells in the biopsy and resection specimen initially suggested the diagnosis of a carcinoid tumor. However, the thick-walled blood vessels, the strong straining for smooth muscle actin and type IV collagen, and the lack of staining for chromogranin and cytokeratin supported the diagnosis of a glomus tumor and militated against a carcinoid tumor. These findings are supported by various immunohistochemical studies in which glomus tumors stain for actin and type IV collagen, and do not stain with cytokeratin or chromogranin. Endoscopic examination remains one of the most reliable means of establishing the diagnosis of a tracheal neoplasm and it should be employed in a patient with a normal chest roentgenogram who has an unexplained cough, hemoptysis, or dyspnea. Glomus tumors in this site behave in a benign way, with complete cure following proper resection. In 4 of 10 reported patients, the tumor extended outside the tracheal wall; therefore, tracheal resection rather than endoscopic therapy should be performed.
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References
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Accepted for publication December 24, 1999.
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Abstract
Introduction
