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Postgraduate Education Corner: Pulmonary, Critical Care, and Sleep Pearls |

A 63-Year-Old Man With a Chronic Cough and an Endobronchial LesionChronic Cough and Endobronchial Lesion FREE TO VIEW

Karim El-Kersh, MD; Rafael L. Perez, MD; Umair Gauhar, MD
Author and Funding Information

From the Department of Pulmonary, Critical Care, and Sleep Disorders Medicine, University of Louisville, Louisville, KY.

Correspondence to: Karim El-Kersh, MD, Department of Pulmonary, Critical Care, and Sleep Disorders Medicine, Ambulatory Care Bldg, 550 S Jackson St, Louisville, KY 40202; e-mail: karimelkersh@yahoo.com


Reproduction of this article is prohibited without written permission from the American College of Chest Physicians. See online for more details.


Chest. 2014;145(4):919-922. doi:10.1378/chest.13-1965
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A 63-year-old man with an extensive smoking history presented with a complaint of persistent dry cough for 3 months. His past medical history was significant for severe COPD with an FEV1 at 40% predicted (stage III by GOLD [Global Initiative for Obstructive Lung Disease] criteria). The patient was retired and a current cigarette smoker, and he had no pets. There was no history of prior malignancy or recent travels. He had no fever, night sweats, or weight loss. He was not receiving any angiotensin-converting enzyme inhibitors. There were no symptoms suggestive of upper airway cough syndrome or gastroesophageal reflux disease.

Physical Examination Findings

The patient was afebrile with stable vital signs. BP was 127/87 mm Hg, heart rate was 85 beats/min, and oxygen saturation was 94% on room air. A complete physical examination was remarkable for yellow nicotine stains on the fingernails without clubbing, wide subcostal angle, distant heart sounds, and prolonged expiratory phase bilaterally, with left-sided monophasic expiratory wheezing best heard over the left upper lobe.

Diagnostic Studies

CBC was normal, apart from a normocytic anemia, with hemoglobin level of 12 g/dL (normal, 14-18 g/dL). Complete metabolic panel results, including electrolytes, renal, and liver function testing, were within normal limits. No acute abnormalities were detected on chest radiograph. Because of the presence of localized wheezing on physical examination, an endobronchial lesion was suspected, and a CT scan of the chest was obtained. The chest CT scan showed emphysematous changes with an upper lobe predominance and an ovoid low-density endobronchial lesion in the distal left mainstem bronchus measuring 2.3 cm in maximum diameter (Fig 1).

Figure Jump LinkFigure 1. CT scan of the chest shows ovoid low-density lesion within the distal left mainstem bronchus near the bifurcation (arrow).Grahic Jump Location
What is the next diagnostic step?
What is the diagnosis?
Next diagnostic step: Bronchoscopy
Diagnosis: Endobronchial hamartoma

Tracheobronchial tumors are rare, accounting for only about 0.4% of all body tumors. In adults, only 10% of the tracheobronchial tumors are benign. Pulmonary hamartomas are the most common benign lung tumors, with an incidence between 0.025% and 0.32%. Most of the pulmonary hamartomas are diagnosed during adulthood, with a peak incidence in the sixth to seventh decade and a male predominance by a ratio of 2:1 to 3:1.

Endobronchial hamartomas (EHs) represent only 1.4% of pulmonary hamartomas, although some studies have reported a higher incidence between 10% and 20%. The term “hamartoma” was coined by Albrecht in 1904 to describe lesions composed of disorganized tissue elements that are native to the organ in which they grow.

EHs can have variable presentations depending on the site and size of the tumor. The patient can be asymptomatic, especially in early stages, but later the patient can develop symptoms of airway irritation or obstruction. Airway irritation can cause symptoms ranging from persistent cough, as in our case, to frank hemoptysis. Airway obstruction can present as dyspnea, atelectasis, and recurrent pneumonias.

Findings on chest CT can be variable because of the various tissue components of EHs. Features such as popcorn calcification that is characteristic of chondroid calcifications or internal fat help to distinguish hamartomas from malignancies. EHs may contain focal collections of fat, which has low attenuation on CT imaging, or collections of fat alternating with foci of calcification. On PET scan, hamartomas usually show little or no uptake but can occasionally be hypermetabolic, mimicking malignant tumors.

The presence of findings suggestive of EH on chest CT scan warrant bronchoscopy as the next diagnostic step. On bronchoscopic examination, EH appears as a well-circumscribed polypoid lesion, either sessile or pedunculated, with a yellowish smooth surface without signs of submucosal infiltration, whereas carcinoid tumors appear as pink to red lesions because of more vascularity.

Differential diagnosis of EH includes all other airway tumors. Airway tumors include primary malignant tumors, such as squamous cell carcinoma from surface epithelium, adenoid cystic carcinoma from salivary glands, and sarcomas from mesenchyme. Secondary malignant tumors can be from direct invasion by tumors from surrounding structures (more common) or due to hematogenous spread (less common). Most of the benign airway tumors (such as hamartoma, leiomyoma, lipoma, fibroma, and neurogenic tumors) arise from the mesenchymal tissue. A small percentage of benign airway tumors can rarely arise from surface epithelium. These include tumors such as squamous cell papilloma, papillomatosis, pleomorphic adenoma, mucous gland adenoma, and oncocytoma.

EHs contain adipose tissue (more than intraparenchymal hamartomas), cartilage, myxomatous connective tissue, and smooth muscle and may have clefts lined with various types of respiratory epithelium. In the largest review series examining 215 cases of pulmonary hamartomas, there was no evidence of either malignant transformation or an unexplained association with other lung neoplasms.

EHs may bleed or obstruct the airways. Thus, treatment should be offered even in asymptomatic cases. The treatment plan should be individualized for each patient considering the site, size, extent of the tumor, and the patient’s comorbidities. Endoscopic removal of benign airway tumors including EHs is preferred, as it is less invasive and provides satisfactory results. Various modalities can be used for endobronchial resection, such as laser, electrocautery, cryotherapy, and argon plasma coagulation. The endoscopic intervention can be provided via flexible or rigid bronchoscopy depending on the size and complexity of the lesion. Treatment directed at the site of the tumor base reduces the chances of local recurrence. Possible complications from such interventions can include bleeding, perforation, and fistula formation. Nonendoscopic surgical intervention ranging from video-assisted thoracoscopic surgery to open thoracotomy should be reserved only for cases not amenable to endobronchial resection. Surgical interventions may include sleeve resection with bronchoplasty, lobectomy, and even pneumonectomy in complicated cases. Nonendoscopic surgeries are also indicated in cases of irreversible lung damage distal to the site of obstruction secondary to longstanding airway obstruction and recurrent infections.

Clinical Course

Based on the findings of low density of the endobronchial lesion on chest CT scan, consideration was given to benign causes, such as EH or lipoma. Thus, the decision was made to perform flexible video bronchoscopy as the next diagnostic and therapeutic step. Bronchoscopic examination showed a whitish, smooth, mobile, polypoid lesion with a narrow stalk causing near complete obstruction of the distal left mainstem bronchus (Fig 2A). The lesion was removed piecemeal using electrocautery snare in blend mode after injecting epinephrine (1:10,000 dilution) locally in the body and stalk of the tumor to prevent bleeding (Fig 2B). The remaining stump of the tumor was coagulated by argon plasma coagulation using 30 W current and 1 L/min flow rate.

Figure Jump LinkFigure 2. A, Polypoid lesion in the distal left mainstem bronchus with near complete obstruction of the lumen. B, Patent airways distal to the lesion after resection using electrocautery snare. Arrow points to the lesion stump close to the left upper lobe bronchus.Grahic Jump Location

Pathologic examination of sections of the resected lesion showed ciliated columnar epithelium covering stroma composed of mature adipose tissue, myxoid areas, smooth muscle bundles, fibrosis, seromucinous glands, and lymphoid aggregates (Fig 3). These findings were consistent with the diagnosis of hamartoma.

Figure Jump LinkFigure 3. A, Hematoxylin and eosin (H&E) stain of one section of the lesion (original magnification × 10). B, Nodule covered by columnar ciliated epithelium (arrowhead), two epithelium-lined clefts (black arrow), and lymphoplasmacytic aggregate (white arrow) (H&E, original magnification × 20). C, Mature adipose tissue (arrow) (H&E, original magnification × 10). D, Smooth muscle hyperplasia (arrow) (H&E, original magnification × 20). E, Myxoid stroma (arrow) (H&E, original magnification × 10). F, Seromucinous glands (arrow) (H&E, original magnification × 20).Grahic Jump Location

Airway patency was restored after the resection. On a follow-up visit 1 month after the procedure, there was marked improvement of the patient’s cough.

  • 1. EH is a rare tumor of the tracheobronchial tree that can have variable presentations, including persistent cough, dyspnea, hemoptysis, and recurrent pneumonias.

  • 2. EHs and lipomas are the main differential diagnosis of an endobronchial lesion with a high fat content on chest CT scan.

  • 3. Endoscopic intervention is the first line of treatment of uncomplicated EHs.

Financial/nonfinancial disclosures: The authors have reported to CHEST that no potential conflicts of interest exist with any companies/organizations whose products or services may be discussed in this article.

Other contributions:CHEST worked with the authors to ensure that the Journal policies on patient consent to report information were met.

Gjevre JA, Myers JL, Prakash UB. Pulmonary hamartomas. Mayo Clin Proc. 1996;71(1):14-20. [CrossRef]
 
Cosío BG, Villena V, Echave-Sustaeta J, et al. Endobronchial hamartoma. Chest. 2002;122(1):202-205. [CrossRef]
 
Lien YC, Hsu HS, Li WY, et al. Pulmonary hamartoma. J Chin Med Assoc. 2004;67(1):21-26.
 
Park CM, Goo JM, Lee HJ, Kim MA, Lee CH, Kang MJ. Tumors in the tracheobronchial tree: CT and FDG PET features. Radiographics. 2009;29(1):55-71. [CrossRef]
 
Mondello B, Lentini S, Buda C, et al. Giant endobronchial hamartoma resected by fiberoptic bronchoscopy electrosurgical snaring. J Cardiothorac Surg. 2011;6:97. [CrossRef]
 
Rodrigues AJ, Coelho D, Dias Júnior SA, Jacomelli M, Scordamaglio PR, Figueiredo VR. Minimally invasive bronchoscopic resection of benign tumors of the bronchi. J Bras Pneumol. 2011;37(6):796-800. [CrossRef]
 

Figures

Figure Jump LinkFigure 1. CT scan of the chest shows ovoid low-density lesion within the distal left mainstem bronchus near the bifurcation (arrow).Grahic Jump Location
Figure Jump LinkFigure 2. A, Polypoid lesion in the distal left mainstem bronchus with near complete obstruction of the lumen. B, Patent airways distal to the lesion after resection using electrocautery snare. Arrow points to the lesion stump close to the left upper lobe bronchus.Grahic Jump Location
Figure Jump LinkFigure 3. A, Hematoxylin and eosin (H&E) stain of one section of the lesion (original magnification × 10). B, Nodule covered by columnar ciliated epithelium (arrowhead), two epithelium-lined clefts (black arrow), and lymphoplasmacytic aggregate (white arrow) (H&E, original magnification × 20). C, Mature adipose tissue (arrow) (H&E, original magnification × 10). D, Smooth muscle hyperplasia (arrow) (H&E, original magnification × 20). E, Myxoid stroma (arrow) (H&E, original magnification × 10). F, Seromucinous glands (arrow) (H&E, original magnification × 20).Grahic Jump Location

Tables

Suggested Readings

Gjevre JA, Myers JL, Prakash UB. Pulmonary hamartomas. Mayo Clin Proc. 1996;71(1):14-20. [CrossRef]
 
Cosío BG, Villena V, Echave-Sustaeta J, et al. Endobronchial hamartoma. Chest. 2002;122(1):202-205. [CrossRef]
 
Lien YC, Hsu HS, Li WY, et al. Pulmonary hamartoma. J Chin Med Assoc. 2004;67(1):21-26.
 
Park CM, Goo JM, Lee HJ, Kim MA, Lee CH, Kang MJ. Tumors in the tracheobronchial tree: CT and FDG PET features. Radiographics. 2009;29(1):55-71. [CrossRef]
 
Mondello B, Lentini S, Buda C, et al. Giant endobronchial hamartoma resected by fiberoptic bronchoscopy electrosurgical snaring. J Cardiothorac Surg. 2011;6:97. [CrossRef]
 
Rodrigues AJ, Coelho D, Dias Júnior SA, Jacomelli M, Scordamaglio PR, Figueiredo VR. Minimally invasive bronchoscopic resection of benign tumors of the bronchi. J Bras Pneumol. 2011;37(6):796-800. [CrossRef]
 
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