*From the Department of Medicine (Drs. Reddy, Govert, and Wahidi), Division of Pulmonary, Allergy and Critical Care Medicine, and the Department of Pathology (Dr. Sporn), Duke University Medical Center, Durham, NC.
Correspondence to: Anita J. Reddy, MD, Associate Staff, Department of Pulmonary, Allergy, and Critical Care Medicine, Cleveland Clinic Health System, 9500 Euclid Ave, Desk A90, Cleveland, OH 44195; e-mail: firstname.lastname@example.org
Pulmonary broncholithiasis can cause a management dilemma depending on its location and the possible involvement of vascular structures. Many patients undergo rigid bronchoscopy or surgical interventions for the removal of broncholiths. In this case report, we describe a 38-year-old white man with a history of performing warehouse demolitions who presented with chronic cough, dyspnea on exertion, and recurrent pneumonia. Imaging studies revealed hilar and mediastinal calcifications, as well as a calcification in the right middle lobe bronchus. Flexible bronchoscopy revealed a mobile obstructing calcified mass in the right middle lobe bronchus. Attempts at removing the mass with forceps were unsuccessful. Instead, the mass was removed using cryotherapy with minimal bleeding and complete resolution of the obstruction. Pathologic examination confirmed that the mass was a broncholith, and stains revealed the presence of histoplasma fungal forms. Partially attached broncholiths can be removed safely using flexible bronchoscopy with the aid of cryotherapy.
A 38-year-old male construction worker was referred to our institution for recurrent pneumonias and right middle lobe obstruction. He had spent most of his life in Texas employed in construction and sheet metal fabrication, but he had spent a year in the Lexington, KY, area performing demolition work.
Seven months prior to presentation at our institution, the patient reported symptoms of persistent dry cough, which then progressed to a cough productive of purulent sputum and dyspnea on exertion. He was treated with antibacterial therapy for right middle lobe pneumonia.
Follow-up chest radiographs revealed persistent right middle lobe infiltrates; therefore, a chest CT scan was performed, revealing multiple calcified hilar and mediastinal lymph nodes, as well as obstruction of the right middle lobe bronchus in association with an area of calcification. Bronchoscopy was performed at the referring institution and revealed an obstruction of the right middle orifice by a mass that could not be removed. Endobronchial biopsies were performed, and pathology findings revealed significant calcium deposits.
Symptoms of fever and productive cough again developed in the patient, and, due to his recurrent symptoms, he was referred to our institution for further evaluation. At the time of presentation to our clinic, he reported mild dry cough without hemoptysis and mild dyspnea on exertion. The findings of the physical examination were normal with normal oxygen saturation while breathing room air. A lung examination did not reveal any bronchial breath sounds, crackles, or wheezes. A chest CT scan (Fig 1
) revealed marked calcification of hilar lymph nodes and the subcarinal lymph node, and an area of dense calcification obstructing the right middle lobe bronchus leading to near-complete collapse of the medial and lateral segments of the right middle lobe. Calcifications were also noted in the spleen.
Repeat bronchoscopy at our institution revealed complete obstruction of the right middle lobe bronchus by a calcified mass, which was partially mobile within the bronchus (Fig 2
). Attempts at removing the mass using forceps were unsuccessful. Cryotherapy was then employed as a means of removal. This resulted in near-complete removal of the mass with resolution of the obstruction, although a small remnant stalk continued to be present within the lateral wall of the right middle lobe bronchus. Coagulation of the stalk was not performed prior to cryotherapy since the stalk was not fully visualized prior to broncholith removal. Minimal bleeding was encountered after removal.
Pathologic examination revealed fragments of chronically inflamed bronchial tissue with fibrocalcific debris consistent with a broncholith (Fig 3
). Histochemical stains were positive for yeast, with morphology typical of Histoplasma capsulatum.
This patient will be followed up for the recurrence of respiratory symptoms. If his cough should return, a follow-up bronchoscopy would be performed to detect the recurrence of broncholithiasis. A chest CT scan would also be considered to evaluate for associated complications of histoplasmosis such as granulomatous mediastinitis or progressive mediastinal fibrosis. Histoplasma is commonly cultured from calcified lymph nodes and broncholiths in asymptomatic patients, and no antimicrobial treatment is recommended unless evidence of chronic histoplasmosis or other complications such as granulomatous mediastinitis develops in the patient.1
Broncholithiasis is a rare disorder that is marked by hilar and/or parenchymal calcifications in response to an inflammatory process. Pulmonary tuberculosis is the most common cause of broncholithiasis worldwide, while histoplasmosis is the most common cause in the United States. Other causes include cryptococcus, coccidioidomycosis, actinomycosis, aspergillosis, nocardiosis, silicosis, and malignancy.
Patients with broncholithiasis can present with a variety of symptoms. The most common symptoms include persistent cough, hemoptysis, recurrent pneumonia, wheezing, and dyspnea. Lithoptysis, or expectoration of small stones, is a rare but pathognomonic sign of broncholithiasis. Broncholithiasis can result in a number of complications, including erosion of broncholiths into the bronchial wall, resulting in airway obstruction and/or hemoptysis, as well as erosion into the esophagus or vasculature.
Chest radiographs can reveal calcification of the hilar and mediastinal lymph nodes, parenchymal calcification, or obstruction of the airways leading to atelectasis, mucoid impaction, bronchiectasis, or airtrapping. Other manifestations include migration or disappearance of a calcific focus.
The management of broncholithiasis is dependent on the degree of symptoms as well as the size and location of the broncholiths. Possible interventions include flexible bronchoscopy, rigid bronchoscopy, and surgical management. Utilizing bronchoscopy for the removal of broncholiths can be difficult due to the embedding of the stone into surrounding structures, large size, and difficulty crushing the stone.
Flexible bronchoscopy can be used to establish a diagnosis, as well as for the removal of broncholiths. Success rates using flexible bronchoscopy for the removal of broncholiths is reported to be approximately 30%, and there have been reports2–4 utilizing balloon catheters and tripod forceps with success, especially in cases where a loose broncholith is present. Complications can include hemoptysis and central airway obstruction due to a loose stone.
Rigid bronchoscopy is also employed for the removal of broncholiths with success rates ranging from 67 to 87%.5 Several institutions have described the use of Nd-YAG and holmium-YAG lasers to fragment large endobronchial stones.3,5 Complications associated with lasers include perforation of the bronchial wall and hemorrhage.
A key question in the management of broncholiths is whether to attempt bronchoscopic removal or to proceed with surgical resection. One large retrospective series by Olsen et al4 identified the degree of attachment of the broncholith to the bronchial wall as a factor in the success rate of bronchoscopic removal. Bronchoscopic excision was successful in 48% of patients with partially eroding broncholiths and in 100% of those with loose broncholiths.
Surgical treatment should be considered over bronchoscopic removal when patients present with hemoptysis, the broncholith is firmly attached to the bronchial wall, or bronchoscopic excision is not available or feasible. Preferred operative procedures include segmentectomy, followed by lobectomy and pneumonectomy.6–7 Complications include laceration of the pulmonary artery, esophageal perforation, and mainstem bronchus tear.
In this case, we describe the first report of using cryotherapy during flexible bronchoscopy for the removal of a broncholith. Cryotherapy has been used for the removal of foreign objects, blood clots, granulation tissue, and mucous plugs,8as well as the management of endobronchial obstruction.9 The advantages of cryotherapy include ease of use, lower cost compared to laser therapies, and reusability of the cryoprobe after disinfection. Complications can include bleeding or airway perforation, especially when intervention includes manipulation of the broncholith stalk. Caution should also be taken when using cryotherapy for this indication and should only be performed by experienced pulmonologists who have been trained in interventional bronchoscopy until further experience is gained and safety is established.
Our experience in this patient indicates that cryotherapy can be an alternative treatment option in the removal of mobile or partially attached broncholiths with flexible bronchoscopy. This modality can be particularly useful when broncholiths are not easily amenable to removal with forceps extraction during flexible bronchoscopy.
The authors have reported to the ACCP that no significant conflicts of interest exist with any companies/organizations whose products or services may be discussed in this article.
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