Ann Thorac Surg 2004;78:2163-2165
© 2004 The Society of Thoracic Surgeons
Case report
Leiomyoma of an Accessory Tracheal Bronchus
Masaya Tamura, MD*,a,
Tomomi Murata, MDa,
Hiroshi Kurumaya, MDa,
Yasuhiko Ohta, MDa
a Departments of Thoracic Surgery and Pathology, Ishikawa Prefectural Central Hospital, Kanazawa, Japan
Accepted for publication August 1, 2003.
* Address reprint requests to Dr Tamura, Department of General and Cardiothoracic Surgery, Kanazawa University School of Medicine, Takaramachi 13-1, Kanazawa, Ishikawa 920-8640, Japan
m-tamura{at}sf.m.kanazawa-u.ac.jp
 |
Abstract
|
|---|
We present a rare case of leiomyoma arising in an accessory right bronchus in a 55-year-old man who had been treated for bronchial asthma. Diagnosis was established on the results of bronchoscopic biopsy. An emergency operation was performed because the patient had begun to complain of severe dyspnea. The tumor was completely removed by electrocautery with a wire snare by rigid bronchoscopy. Postoperative bronchoscopic findings revealed a tracheal bronchus arising above the carina, and the root of the tumor existed in the orifice of the branch. Final histology revealed a leiomyoma.
|
Introduction
|
|---|
Tracheal bronchus is a rare anomaly in which a bronchus arises directly from the trachea; this is a common finding in sheep, swine, cattle, camels, goat, and giraffes, but it is a rare and usually incidental finding in humans. Most tumors of the tracheobronchial tree are malignant in nature, and benign tumors are quite rare [1]. We believe this report is the first case of leiomyoma arising from an accessory tracheal bronchus.
A 55-year-old man presented with a history of bronchial asthma for 8 years and two episodes of pneumonia in the previous 6 months. The patient had a complaint of mild dyspnea. Computed tomography showed a 2 cm fat-density mass obstructing the lumen of the trachea. The opening of the tracheal bronchus was also seen on chest computed tomography (Fig 1A). At bronchoscopy, a smooth, round mass was seen in the trachea, arising from the right tracheal wall and obstructing the view of the trachea (Fig 1B). Biopsy was easily taken and histology revealed a leiomyoma. An emergency operation was scheduled 5 days after the biopsy because the patient had begun to complain of severe dyspnea. Electrocautery with a wire snare was applied using rigid bronchoscopy to remove the polypoid lesion under general anesthesia with a percutaneous cardiopulmonary support system for hypoxemia during the operation. The base of the tumor was treated with bronchoscopic neodymium-yttrium-aluminum-garnet laser, because frozen section diagnosis was not available late at night. The resected specimen was an ovoid encapsulated mass measuring 1.9 x 1.6 cm. Pathology confirmed the diagnosis of leiomyoma. No mitosis, necrosis, or nuclear hyperchromasia was observed, and the resection margins were tumor free. Postoperative bronchoscopic image showed the opening of the tracheal bronchus and the root of the tumor existed in the orifice of the branch (Fig 2). The patient is well with no evidence of recurrence of tumor and no occurrence of pneumonia 6 months postoperatively.
View larger version (64K):
[in this window]
[in a new window]
|
Fig 1. (A) Chest computed tomography depicting a round polypoid tumor and the accessory tracheal bronchus originating at the right wall of the lower trachea. (B) Endoscopic findings reveal a polypoid tumor with smooth surface occupying approximately 90% of the trachea.
|
|
View larger version (72K):
[in this window]
[in a new window]
|
Fig 2. (A) Postoperative bronchoscopic findings showed an accessory opening in the right wall of the trachea 1 cm above the carina (small arrows) and also reveals the carina (large arrow). (B) The root of the tumor existed in the orifice of the tracheal bronchus.
|
|
|
Comment
|
|---|
Benign tumors of the trachea are rare, and reports of leiomyoma are even less common. Grillo and Mathisen [1] reported that tracheal leiomyoma accounts for approximately 1% of all tracheal tumors. As in the present case, many patients are initially diagnosed with asthma or chronic bronchitis, which may delay the correct diagnosis. Tracheal tumor should be considered in patients who present sudden onset of asthma, no response to medication, and change of symptoms according to posture.
A tracheal bronchus is an aberrant, most commonly ectopic, or accessory bronchus arising almost invariably from the right lateral wall of the trachea, usually within 2 cm above the carina (range of incidence, 0.1% to 0.6%). The diameter ranges from 0.5 to 1.0 cm, and the length ranges from 0.6 to 2.0 cm [2]. Most cases of tracheal bronchus are asymptomatic, but some patients may experience recurrent pneumonia, chronic bronchitis, or bronchiectasis [3]. Our patient suffered pneumonia of the right upper lobe. This may have been caused by poor local drainage of the involved bronchi. Although it is difficult to determine which one of the two causes, leiomyoma or tracheal bronchus, led to recurrent pneumonia, the patient has been free of any disease for 6 postoperative months. Careful follow-up should be needed, and if recurrent pneumonia is related to the tracheal bronchus, surgical treatment of this bronchus may be an indication. Tracheal bronchus can be identified during bronchoscopic examination by the presence of an ectopic opening from the tracheal wall. We could not detect the anomaly of the trachea in the preoperative bronchoscopy because of the presence of the tumor obscuring it. Computed tomography is the newest modality for evaluating tracheal bronchus. In our case, the opening of the tracheal bronchus was seen on the preoperative chest computed tomographic scan. Several cases of anomalies of the bronchi combined with lung carcinoma [4, 5] and carcinoid tumor [6] were identified in the literature, but none of them were tracheal leiomyoma.
Recently a variety of techniques have been used in treating tracheal tumors, such as neodymium-yttrium-aluminum-garnet laser and electrosurgery with fiberoptic bronchoscope and snare. These procedures are especially useful for the pedunculated polyp. In our case, tumor resection was performed using rigid bronchoscope under general anesthesia with a percutaneous cardiopulmonary support system for the following reasons: the patient was under hypoxia (SaO2 was 
89% to 92% under room air) preoperatively, and we were afraid of suffocation because the tumor existed just above the carina.
Recurrence of tracheal leiomyoma is rare with only 2 reported cases in the literature. One of these was incompletely removed by bronchoscopy and recurred within 7 months [7]. The other was removed by segmental sleeve resection with end-to-end anastomosis; this mass recurred 12 years later in the same area [8]. The second case was unusual because it also presented with a calcified extratracheal extension of the leiomyoma. In our case, we performed additional treatment on the base of the tumor with neodymium-yttrium-aluminum-garnet laser because frozen section examination was not available late at night for the emergency operation.
Conservative treatment for leiomyoma of the trachea should be selected depending on the location and size of the tumor, and careful follow- up should be done, keeping in mind both the possibility of recurring tumor and pneumonia.
|
References
|
|---|
- Grillo HC, Mathisen J. Primary tracheal tumors: treatment and results. Ann Thorac Surg. 1990;49:69–77[Abstract]
- Barat M, Konrad HR. Tracheal bronchus. Am J Otolaryngol. 1987;8:118–122[Medline]
- Oísullivan BP, Frassica JJ, Rayder SM. Tracheal bronchus: a cause of prolonged atelectasis in intubated children. Chest. 1998;113:537–540[Abstract/Free Full Text]
- Sato M, Hasegawa S, Shoji T, Wada H. Tracheobronchoplasty for resection of lung cancer arising from a tracheal bronchus. Ann Thorac Surg. 2002;73:310–312[Abstract/Free Full Text]
- Kuo CW, Lee YC, Perng RP. Tracheal bronchus associated with lung cancer. A case report. Chest. 1999;116:1125–1127[Abstract/Free Full Text]
- Patrinou V, Kourea H, Dougenis D. Bronchial carcinoid of an accessory tracheal bronchus. Ann Thorac Surg. 2001;71:1034–1035[Abstract/Free Full Text]
- Harris PF, Maness MD, Ward PH. Leiomyoma of the larynx and trachea. South Med J. 1967;60:1223–1227[Medline]
- Rodriquez E, Pombo F, Aguilera C, Capdevila A. Recurring tracheal leiomyoma presenting as a calcified mediastinal mass. Eur J Radiol. 1996;22:82–84
This article has been cited by other articles:
|
|
|
|
 
Radiology Quiz Case: Diagnosis
Arch Otolaryngol Head Neck Surg,
April 1, 2006;
132(4):
454 - 454.
[Full Text]
[PDF]
|
|
|
Top
Abstract
Introduction
