To the Editor:
Endoscopic resection of tracheal and bronchial tumors has recently gained a wider acceptance among the interventional pulmonology and thoracic surgery community, as a minimally invasive treatment modality offering the benefit of lung function preservation. Although the application of this intervention in malignant tumors is mainly palliative in nature, there remain concerns regarding the medium and long-term outcomes and the risk of local recurrence in benign and low-grade malignant tumors.
We describe the case of a 40-year-old female nurse who presented with wheezing and complaints of dyspnea. She was treated for asthma, but after 2 episodes of recurrent pneumonia, underwent computed tomography of the chest that demonstrated a 2.9×1.6 cm tumor in the left main stem bronchus (Fig. 1A). Flexible bronchoscopy revealed a wide based tumor attached to the membranous airway, and occluding >90% of the left main stem bronchus (Fig. 1B). Incisional biopsy with histologic and immunohistochemical staining revealed desmin-positive cells and no atypia, increased mitosis or necrosis (Figs. 1C, D). These findings were suggestive of benign spindle cell proliferation and very suspicious for leiomyoma.
The decision was made to avoid a pneumonectomy or sleeve pneumonectomy, in a young and otherwise healthy patient, and to proceed with endoscopic resection.
In the operating room, the patient was placed under general anesthesia and intubated with a 11 to 12-mm rigid bronchoscope, through which jet ventilation was established (Karl Storz, Tuttlingen, BW, Germany). Using a combination of the holmium: yttrium aluminum garnet (YAG) at the setting of 0.2 J and 10 to 20 Hz, and neodymium-doped:YAG laser at the setting of 20 W and 0.5-second pulse, the tumor was completely resected in 1 session. This resulted in near complete opening of the left main stem bronchus. Clinically, the patient became completely asymptomatic and had no episode of hemoptysis after the procedure. Endoscopic follow-up with biopsy at 1, 6, 12, and 24 months showed no residual disease or evidence of tumor recurrence.
Primary leiomyomas of the lung are extremely rare, with the majority (51%) occurring in the parenchyma, and the rest either endobronchial (33%) or within the trachea (16%).1 These tumors usually present in the adult population with a slight female preponderance. Literature is very limited, with only 108 cases of endobronchial leiomyomas reported in a recent review.1 These patients commonly present with symptoms of partial or complete airway obstruction, such as cough, wheezing, shortness of breath, dyspnea, and chest pain. Recurrent post obstructive focal pneumonitis can occur in the affected bronchus, and hemoptysis may be seen with ulceration of the tumor surface.
Radiographically, atelectasis or consolidation from focal pneumonitis of the affected lung segment can be seen. Computed tomography will delineate the size, location, and usual round configuration of these tumors, which are otherwise seldom calcified.2
Definitive diagnosis is made with bronchoscopy, which allows for direct visualization and tissue sampling for histologic analysis. These tumors are generally polypoid with a wide base, as in our patient. Histologic criteria for characterization include cellularity, mitotic activity, and pleiomorphic characteristics. Mitotic activity is one of the main determinants in excluding malignancy, and positive immunohistochemical staining for desmin usually supports the diagnosis of leiomyoma.2 Malignant transformation of these tumors has yet to be reported in the literature.
Removal is largely based on treating the patient’s symptoms. Surgical excision of these benign tumors was historically achieved with lobectomy, segmentectomy, pneumonectomy, or even tracheal resection. Less radical surgical approaches include bronchotomy with bronchoplasty, and sleeve resection of the involved bronchus. More conservative approaches with bronchoscopic resection have been recently described, particularly with smaller and more proximal tumors, in a lung parenchyma preserving effort.3 The endoscopic technique depends on the shape and location of the tumor, and includes snaring, electrocautery, and laser, as we describe in this manuscript. These endoscopic modalities offer the advantage of being less invasive while preserving the lung function. Our patient was in fact discharged home the next day and had complete symptomatic relief. The properties of the laser technology allow safe and accurate endoscopic resection in view of the depth of penetration of the holmium YAG (5 mm), and the ability to achieve excellent hemostasis.4 Despite good short and medium-term outcomes, local recurrence may still occur after any treatment modality, especially endoscopic resection.3 Risk factors for local recurrence include a wide tumor base, as shown by Park et al.5 Therefore, the authors believe lifetime bronchoscopic surveillance is still warranted to ensure absence of recurrence.
Charles Bakhos, MD*
Costas Stavrakis, MD†
Michael Presta, MD‡
*Department of Thoracic Medicine and Surgery, Lewis Katz School of Medicine, Temple University Hospital Philadelphia, PA
Departments of †Radiology
‡Pathology, Albany Medical Center Albany, NY
1. Swarnakar R, Sinha S. Endobronchial leiomyoma: a rare and innocent tumour of the bronchial tree. Lung India. 2013;30:57–60.
2. Kim YK, Kim H, Lee KS, et al. Airway leiomyoma: imaging findings and histopathologic comparisons in 13 patients. Am J Roentgenol. 2007;189:393–399.
3. Kwon YS, Kim H, Koh WJ, et al. Clinical characteristics and efficacy of bronchoscopic intervention for tracheobronchial leiomyoma. Respirology. 2008;13:908–912.
4. Mohan A, Guleria R, Mohan C, et al. Laser bronchoscopy: current status. J Assoc Physicians India. 2004;52:915–920.
5. Park JS, Lee M, Kim HK, et al. Primary leiomyoma of the trachea, bronchus, and pulmonary parenchyma—a single-institutional experience. Eur J Cardiothorac Surg. 2012;41:41–45.