Ann Thorac Surg 2005;79:2127-2128
© 2005 The Society of Thoracic Surgeons
Case report
Resection of the Entire Left Mainstem Bronchus for an Inflammatory Pseudotumor
Robert J. Cerfolio, MD, FACS,
Thomas C. Matthews, BS*
Department of Cardiothoracic Surgery, University of Alabama School of Medicine, Birmingham, Alabama
Accepted for publication November 25, 2003.
* Address reprint requests to Dr Matthews, University of Alabama School of Medicine, 1900 University Blvd, THT Room 712, Birmingham, AL35294-0016 (E-mail: tcm78{at}uab.edu).
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Abstract
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We present the case of a 16-year-old white girl with a history of recurrent postobstructive pneumonia. Chest roentgenogram, chest computed tomography, and bronchoscopy revealed a mass in the left mainstem bronchus with an exophytic component. Multiple bronchoscopic biopsies confirmed the mass to be an inflammatory pseudotumor. After failing months of medical therapy with systemic steroids as well as several laser ablations, the tumor was removed through a left thoracotomy with resection of the entire left mainstem bronchus and reimplantation of the left upper and lower lobe into the trachea without complication or recurrence after 1 year.
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Introduction
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Inflammatory pseudotumors of the lung are poorly defined pulmonary lesions. Although these pseudotumors are quite rare, they are the most common primary lung neoplasm in the pediatric population [1]. The majority of these pseudotumors are located in the lung parenchyma, but 5% to 16% are endobronchial in location [2]. Clinically, inflammatory pseudotumors are difficult lesions to diagnose. Tissue diagnosis is often unreliable from fine needle aspiration or frozen tissue section, or both. Although medical management is often tried and is sometimes successful, surgical extirpation is usually required [3]. We present an unusual case of an endobronchial inflammatory pseudotumor of the entire left mainstem bronchus in a young female.
A 16-year-old white girl presented with fever, left-sided chest pain, and dyspnea at rest. Her past medical history was significant for recurrent bronchitis and left-sided postobstructive pneumonia. Past workup included a computed tomographic scan of the chest (Fig 1),which demonstrated a large endobronchial abnormality in and around the left mainstem bronchus near the carina. Multiple biopsies of the mass by bronchoscopy showed an inflammatory pseudotumor of the lung. The patient underwent 9 months of rigid bronchoscopy with laser therapy and dilations as well as systemic steroids. However, she continued to re-obstruct, and her symptoms recurred. Repeat computed tomography on this admission revealed the known mass and consolidation of the entire left lung with associated volume loss. The location was confirmed on bronchoscopy. The decision was made to remove the tumor through a left thoracotomy.
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Fig 1. Computed tomographic scan of the chest demonstrating exophytic mass in the left mainstem bronchus with distal obstructive pneumonia.
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The patient was brought to the operating room for bronchoscopy followed by a mediastinoscopy. The mediastinoscopy was performed to mobilize the trachea and mainstem bronchi, to evaluate the exophytic nature of the mass, and to determine the extent of carina involvement. Because there appeared to be little inflammatory reaction around the carina or right mainstem bronchus, a left muscle and rib-sparing thoracotomy was performed. The intercostal muscle between the forth and fifth ribs was harvested with cautery, preventing later calcification from residual periosteal tissue. Exploration revealed a chronically atelectatic left lung and multiple adhesions. The ligamentum arteriosum was divided and ligated in order to mobilize the descending thoracic aorta. The aorta was encircled with a wide plastic drain in between the left subclavian and common carotid artery. Another plastic drain was placed around the aorta distal to the left subclavian and retracted cephalad. A third plastic drain was placed under the left mainstem bronchus after freeing it from the pericardium (Fig 2). The left mainstem bronchus was then cut off the trachea flush with the carina. This allowed us to look inside the divided left mainstem, and we noted that the tumor was present in the airway to the airflow divider between the left upper and lower lobes, arising from the membranous portion of the bronchus. The exophytic aspect of this tumor was easily resected from its surrounding structures, and it was a minor component of the mass. A proximal trachea margin was sent for frozen section, which was found to be negative. Because the process involved the entire left mainstem bronchus, a distal margin was cut just at the airflow divider, which was also negative on frozen section. An anastomosis was then performed using 3-0 interrupted PDS (polydioxanone) sutures (Ethicon, Somerville, NJ). The posterior part of the anastomosis on the membranous portion of the airway was run with knots on the outside. The tension free, epithelial to epithelial anastomosis was then completed with interrupted PDS sutures anteriorly, again with knots outside the airway. At the completion of the anastomosis, the left lung was reinflated and there was no anastomotic leak. Then an intraoperative bronchoscopy was performed, which confirmed the adequacy of the anastomosis. The previously harvested intercostal muscle flap devoid of periosteum was then wrapped around the anterior portion of the anastomosis and secured with two interrupted Prolene sutures (Ethicon, Somerville, NJ).
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Fig 2. (A) Wide plastic drains were placed in between the left subclavian and common carotid artery and (B) around the aorta distal to the left subclavian. (C) A third plastic drain was placed under the left mainstem bronchus after freeing it from the pericardium.
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The patient had an uneventful postoperative course and left the hospital on postoperative day 4. The final pathology report described a spindle cell proliferation consistent with the diagnosis of inflammatory pseudotumor, excised in its entirety. The total length of the left main stem bronchus that was removed measured 4.6 cm. Postoperative follow-ups at 2, 9 and 14 months using bronchoscopy and computed tomography demonstrated no evidence of recurrence, satisfactory anastomotic healing, and a well-expanded left lung.
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Comment
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Pulmonary inflammatory pseudotumors are unusual neoplasms that are difficult to characterize pathologically. For this reason they have been referred to by a large number of terms in the literature. For example, they have been called plasma cell granuloma, fibrous histiocytoma, or fibroxanthoma, all terms that describe the microscopic pathologic process [4]. The most current accurate description is a benign pulmonary neoplasm, typically parenchymal in origin, primarily composed of plasma cells and spindle-shaped mesenchymal cells [4]. These tumors comprise less than 1% of all pulmonary tumors and have no predilection for either gender [5]. In the small subset of patients who have endobronchial lesions develop, the presentation may be more acute and clinically impressive due to the postobstructive pneumonia.
Endobronchial inflammatory pseudotumors present a unique opportunity for surgical excision using sleeve techniques. Because the pseudotumor was located on the left side of our patient, surgical resection was more difficult, and thus medical therapy was attempted first. Intraluminal bronchoscopic removal has been discussed; however current data are lacking on the long-term success of these techniques [6]. Because our attempts failed to successfully treat this patient through a medical therapy approach, and because of her age and the size and exophytic nature of the mass, a surgical resection was performed. In addition, because the preoperative diagnosis is often in doubt, surgery remains the best diagnostic method and treatment modality of choice for these rare tumors [4, 5].
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Acknowledgments
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Before performing this operation we received invaluable advice from Doug Woods, MD (Seattle), Eric Vallieres (Seattle) and Dan Miller (Emory). We appreciate their time and expertise.
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References
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- Bahadori M, Liebow AA. Plasma cell granulomas of the lung Cancer 1973;31:191-208.[Medline]
- Hajjar WA, Ashour MH, Al-Rikabi AC. Endobronchial inflammatory pseudotumor of the lung Saudi Med J 2001;22:366-368.[Medline]
- Alexiou C, Obuszko Z, Beggs D, Morgan WE. Inflammatory pseudotumors of the lung Ann Thorac Surg 1998;66:948-950.[Abstract/Free Full Text]
- Cerfolio RJ, Allen MS, Nascimento AG, et al. Inflammatory pseudotumors of the lung Ann Thorac Surg 1999;67:933-936.[Abstract/Free Full Text]
- Copin MC, Gosselin BH, Ribet ME. Plasma cell granuloma of the lungdifficulties in diagnosis and prognosis. Ann Thorac Surg 1996;61:1477-1482.[Abstract/Free Full Text]
- Dahabreh J, Zisis C, Arnogiannaki N, et al. Inflammatory pseudotumora controversial entity. Eur J Cardio-Thorac 1999;16:670-673.
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Abstract
Introduction
