Case Study
Glomus tumor of the trachea Seyyed Hossein Fattahi Masoum1, Amir Hossein Jafarian2, Ali Reza Sharifian Attar3, Davood Attaran4, Reza Afghani5 and Azadeh Jabbari Noghabi6
Asian Cardiovascular & Thoracic Annals 2015, Vol. 23(3) 325–327 ß The Author(s) 2014 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492314528184 aan.sagepub.com
Abstract Glomus tumors of the trachea are rare and benign, but most become symptomatic, so they need intervention. A 21-yearold man was evaluated due to cough and hemoptysis. Computed tomography and bronchoscopy showed a polypoid mass above the carina. The tumor was removed completely by rigid bronchoscopy. The pathologic diagnosis was glomus tumor. After one year, because of recurrence of the tumor at the same site, the patient underwent reoperation, and resection and anastomosis of trachea through a right posterolateral thoracotomy was performed.
Keywords Endoscopy, glomus tumor, tracheal neoplasms
Introduction Glomus tumor is a benign neoplasm that originates from modified smooth muscle cells of the normal glomus body.1,2 This tumor affects primarily the skin and is very rare in the trachea where normal glomus bodies may be absent. Less than 20 cases of these tumors have been reported.1 A tumor in the trachea usually protrudes into the lumen like a polypoid mass, which causes partial obstruction.2 Because of symptoms, intervention is unavoidable. Despite the benign behavior of glomus tumors, recurrence is seen in cases of incomplete resection, so complete resection and anastomosis of the trachea is preferred.3
Case report A 21-year-old man with cough and hemoptysis for some months was referred by a pulmonologist with a diagnosis of tracheal tumor above the carina on computed tomography (Figure 1). We decided to take a biopsy of the tumor through rigid bronchoscopy under general anesthesia. On bronchoscopy, a protruding mass was seen 2 cm above the carina, but the tumor was fragile and we had to discontinue the procedure because of bleeding. After ensuring hemostasis and stable condition, the endotracheal tumor was removed completely through rigid bronchoscopy. The
pathologic finding was glomus tumor of the trachea, which was confirmed immunohistochemically. We recommend surgery, but the patient preferred to wait and see if the tumor recurred. On follow-up after one year, the tumor recurred in the same site, and he underwent reoperation. Resection and end-to-end anastomosis of the trachea was accomplished through a right posterolateral thoracotomy (Figure 2). The postoperative period was uneventful and the patient was discharged home. Follow-up bronchoscopy was performed 2 month later. After one year of follow-up, he was well with no problem. 1 Endoscopic & Minimally Invasive Surgery Research Center, Ghaem Hospital, Mashhad, Iran 2 Department of Pathology, Ghaem Hospital, Mashhad, Iran 3 Department of Anesthesiology, Ghaem Hospital, Mashhad, Iran 4 COPD Research Center, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran 5 Cardiothoracic Surgery & Transplant Research Center, Emam Reza Hospital, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran 6 Department of General Surgery, Emam Reza Hospital, Mashhad University of Medical Sciences, Mashhad, Iran
Corresponding author: Reza Afghani, Thoracic Surgery, Cardiothoracic Surgery & Transplant Research Center, Emam Reza Hospital, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran. Email: [email protected]
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Asian Cardiovascular & Thoracic Annals 23(3)
Discussion Glomus bodies are specialized arteriovenous structures surrounded by modified smooth muscle cells which are called glomus cells. They are most often found in the dermis and subcutaneous tissues and regulate blood flow and temperature through the skin. These structures are sparse or even absent in visceral organs, making them a rare finding in the trachea.4 Most of these tumors are not invasive and extratracheal extension is rare, but they have the potential to grow and protrude into the lumen. This results in symptoms that make intervention necessary. According to previous reports, the most common symptoms of these tumors are shortness of breath, cough, and stridor due to tracheal obstruction, and hemoptysis due to the
vascularity of the tumor.5,6 Glomus tumors can mimic carcinoid tumors that are the main differential diagnosis of glomus tumors. Biopsy of the tumor and immunohistochemistry can differentiate carcinoid from glomus tumors. The distinguishing features are the presence of neuroendocrine differentiation and immunoreactivity for cytokeratin and chromogranin in carcinoid tumors, and positivity for desmin and smooth muscle actin in glomus tumors (Figure 3).7,8 Because they become symptomatic, glomus tumors need intervention. Sometimes, because of stridor and dyspnea, rigid bronchoscopy and removal of the tumor can palliate the symptoms. Rigid bronchoscopy can also help in accurate diagnosis. In some reports, endoscopic removal of the tumor proved to be
Figure 1. Three-dimensional computed tomography with reconstruction of the trachea.
Figure 2. Resection of the trachea at the site of the tracheal glomus tumor through a right posterolateral thoracotomy. A: tumor; B: distal trachea; C: proximal trachea.
Figure 3. Monotonous proliferation of oval cells with clear cells around a dilated vessel. Hematoxylin and eosin stain, original magnification 40.
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adequate therapy, and in cases of frequent recurrence, stenting might be suitable to palliate symptoms.1,3 In some cases, endoscopic resection was accomplished using a ND:YAG laser to ablate the base of the tumor.1 Despite the benign behavior of glomus tumors, they may recur after complete endoscopic removal. Surgical resection of the trachea and reconstruction by end-to-end anastomosis can eradicate these tumors because of their benign nature.3 Overall, it seems that if the patient can tolerate surgery, it is better to offer surgery (resection and anastomosis) for glomus tumor of the trachea, and to use an alternative approach in patients unfit for surgery or who refuse this approach.3 Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
2.
3.
4.
5.
6.
7.
Conflict of interest statement 8.
None declared.
bronchoscopy: a case report. J Thorac Oncol 2008; 9: 1065–1067. Takahashi N, Oizumi H, Yanagawa N and Sadahiro M. A bronchial glomus tumor surgically treated with segmental resection. Interact Cardiovasc Thorac Surg 2006; 5: 258–260. Baek SH, Huh DM, Park JH, Kwak EK, Kim BH and Han WK. Glomangiomyoma of the trachea. Korean J Thorac Cardiovasc Surg 2011; 44: 440–443. Sakr L, Palaniappan R, Payan MJ, Doddoli C and Dutau H. Tracheal glomus tumor: a multidisciplinary approach to management. Respir Care 2011; 56: 342–346. Garcı´ a-Prats MD, Sotelo-Rodrı´ guez MT, Ballestı´ n C, et al. Glomus tumor of the trachea: report of a case with microscopic, ultrastructural immunohistochemical examination and review of the literature. Histopathology 1991; 19: 459–464. Koskinen SK, Niemi PT, Ekfors TO, Sipila J, Valavaara R and Dean PB. Glomus tumor of the trachea. Eur J Radiol 1998; 8: 364–366. Okereke IC, Sheski FD and Cummings OW. Glomus tumor of the trachea. J Thorac Oncol 2011; 6: 1290–1291. Grillo H. Surgery of the Trachea and Bronchi. Hamilton, Ontario: BC Decker, Inc., 2004, pp. 94–95.
References 1. Colaut F, Toniolo L, Scapinello A and Pozzobon M. Tracheal glomus tumor successfully resected with rigid
Downloaded from aan.sagepub.com at UCSF LIBRARY & CKM on April 12, 2015
Glomus tumor of the trachea Seyyed Hossein Fattahi Masoum1, Amir Hossein Jafarian2, Ali Reza Sharifian Attar3, Davood Attaran4, Reza Afghani5 and Azadeh Jabbari Noghabi6
Asian Cardiovascular & Thoracic Annals 2015, Vol. 23(3) 325–327 ß The Author(s) 2014 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492314528184 aan.sagepub.com
Abstract Glomus tumors of the trachea are rare and benign, but most become symptomatic, so they need intervention. A 21-yearold man was evaluated due to cough and hemoptysis. Computed tomography and bronchoscopy showed a polypoid mass above the carina. The tumor was removed completely by rigid bronchoscopy. The pathologic diagnosis was glomus tumor. After one year, because of recurrence of the tumor at the same site, the patient underwent reoperation, and resection and anastomosis of trachea through a right posterolateral thoracotomy was performed.
Keywords Endoscopy, glomus tumor, tracheal neoplasms
Introduction Glomus tumor is a benign neoplasm that originates from modified smooth muscle cells of the normal glomus body.1,2 This tumor affects primarily the skin and is very rare in the trachea where normal glomus bodies may be absent. Less than 20 cases of these tumors have been reported.1 A tumor in the trachea usually protrudes into the lumen like a polypoid mass, which causes partial obstruction.2 Because of symptoms, intervention is unavoidable. Despite the benign behavior of glomus tumors, recurrence is seen in cases of incomplete resection, so complete resection and anastomosis of the trachea is preferred.3
Case report A 21-year-old man with cough and hemoptysis for some months was referred by a pulmonologist with a diagnosis of tracheal tumor above the carina on computed tomography (Figure 1). We decided to take a biopsy of the tumor through rigid bronchoscopy under general anesthesia. On bronchoscopy, a protruding mass was seen 2 cm above the carina, but the tumor was fragile and we had to discontinue the procedure because of bleeding. After ensuring hemostasis and stable condition, the endotracheal tumor was removed completely through rigid bronchoscopy. The
pathologic finding was glomus tumor of the trachea, which was confirmed immunohistochemically. We recommend surgery, but the patient preferred to wait and see if the tumor recurred. On follow-up after one year, the tumor recurred in the same site, and he underwent reoperation. Resection and end-to-end anastomosis of the trachea was accomplished through a right posterolateral thoracotomy (Figure 2). The postoperative period was uneventful and the patient was discharged home. Follow-up bronchoscopy was performed 2 month later. After one year of follow-up, he was well with no problem. 1 Endoscopic & Minimally Invasive Surgery Research Center, Ghaem Hospital, Mashhad, Iran 2 Department of Pathology, Ghaem Hospital, Mashhad, Iran 3 Department of Anesthesiology, Ghaem Hospital, Mashhad, Iran 4 COPD Research Center, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran 5 Cardiothoracic Surgery & Transplant Research Center, Emam Reza Hospital, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran 6 Department of General Surgery, Emam Reza Hospital, Mashhad University of Medical Sciences, Mashhad, Iran
Corresponding author: Reza Afghani, Thoracic Surgery, Cardiothoracic Surgery & Transplant Research Center, Emam Reza Hospital, Faculty of Medicine, Mashhad University of Medical Sciences, Mashhad, Iran. Email: [email protected]
Downloaded from aan.sagepub.com at UCSF LIBRARY & CKM on April 12, 2015
326
Asian Cardiovascular & Thoracic Annals 23(3)
Discussion Glomus bodies are specialized arteriovenous structures surrounded by modified smooth muscle cells which are called glomus cells. They are most often found in the dermis and subcutaneous tissues and regulate blood flow and temperature through the skin. These structures are sparse or even absent in visceral organs, making them a rare finding in the trachea.4 Most of these tumors are not invasive and extratracheal extension is rare, but they have the potential to grow and protrude into the lumen. This results in symptoms that make intervention necessary. According to previous reports, the most common symptoms of these tumors are shortness of breath, cough, and stridor due to tracheal obstruction, and hemoptysis due to the
vascularity of the tumor.5,6 Glomus tumors can mimic carcinoid tumors that are the main differential diagnosis of glomus tumors. Biopsy of the tumor and immunohistochemistry can differentiate carcinoid from glomus tumors. The distinguishing features are the presence of neuroendocrine differentiation and immunoreactivity for cytokeratin and chromogranin in carcinoid tumors, and positivity for desmin and smooth muscle actin in glomus tumors (Figure 3).7,8 Because they become symptomatic, glomus tumors need intervention. Sometimes, because of stridor and dyspnea, rigid bronchoscopy and removal of the tumor can palliate the symptoms. Rigid bronchoscopy can also help in accurate diagnosis. In some reports, endoscopic removal of the tumor proved to be
Figure 1. Three-dimensional computed tomography with reconstruction of the trachea.
Figure 2. Resection of the trachea at the site of the tracheal glomus tumor through a right posterolateral thoracotomy. A: tumor; B: distal trachea; C: proximal trachea.
Figure 3. Monotonous proliferation of oval cells with clear cells around a dilated vessel. Hematoxylin and eosin stain, original magnification 40.
Downloaded from aan.sagepub.com at UCSF LIBRARY & CKM on April 12, 2015
Masoum et al.
327
adequate therapy, and in cases of frequent recurrence, stenting might be suitable to palliate symptoms.1,3 In some cases, endoscopic resection was accomplished using a ND:YAG laser to ablate the base of the tumor.1 Despite the benign behavior of glomus tumors, they may recur after complete endoscopic removal. Surgical resection of the trachea and reconstruction by end-to-end anastomosis can eradicate these tumors because of their benign nature.3 Overall, it seems that if the patient can tolerate surgery, it is better to offer surgery (resection and anastomosis) for glomus tumor of the trachea, and to use an alternative approach in patients unfit for surgery or who refuse this approach.3 Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
2.
3.
4.
5.
6.
7.
Conflict of interest statement 8.
None declared.
bronchoscopy: a case report. J Thorac Oncol 2008; 9: 1065–1067. Takahashi N, Oizumi H, Yanagawa N and Sadahiro M. A bronchial glomus tumor surgically treated with segmental resection. Interact Cardiovasc Thorac Surg 2006; 5: 258–260. Baek SH, Huh DM, Park JH, Kwak EK, Kim BH and Han WK. Glomangiomyoma of the trachea. Korean J Thorac Cardiovasc Surg 2011; 44: 440–443. Sakr L, Palaniappan R, Payan MJ, Doddoli C and Dutau H. Tracheal glomus tumor: a multidisciplinary approach to management. Respir Care 2011; 56: 342–346. Garcı´ a-Prats MD, Sotelo-Rodrı´ guez MT, Ballestı´ n C, et al. Glomus tumor of the trachea: report of a case with microscopic, ultrastructural immunohistochemical examination and review of the literature. Histopathology 1991; 19: 459–464. Koskinen SK, Niemi PT, Ekfors TO, Sipila J, Valavaara R and Dean PB. Glomus tumor of the trachea. Eur J Radiol 1998; 8: 364–366. Okereke IC, Sheski FD and Cummings OW. Glomus tumor of the trachea. J Thorac Oncol 2011; 6: 1290–1291. Grillo H. Surgery of the Trachea and Bronchi. Hamilton, Ontario: BC Decker, Inc., 2004, pp. 94–95.
References 1. Colaut F, Toniolo L, Scapinello A and Pozzobon M. Tracheal glomus tumor successfully resected with rigid
Downloaded from aan.sagepub.com at UCSF LIBRARY & CKM on April 12, 2015
