The patient developed recurrent respiratory tract infection. She was treated for recurrent cough and polypnea. Treatments such as inhalation of steroid hormones and bronchodilators proved ineffective. The preliminary diagnosis revealed a barrier to secretion drainage after the airway cartilage was destroyed by trauma, and pathogenic infection by bacteria and fungi, based on the endoscopic and pathologic findings. However, the nature of “bone-like substances” in the lumen could not be determined. The “bone-like substances” were found to be gray-white and hard in texture when preparing pathologic sections. During staining, most of them chipped off and decalcified, and no tissue structure was observed. Chest CT and bronchoscopy showed it as “broncholithiasis (intraluminal type)” combined with scattered spot-like calcification. Therefore, the stone was removed under a bronchoscope, and oral administration of itraconazole (6.5 mL) was initiated for 2 months for antifungal treatment. The patient had no symptom of cough, polypnea, and so forth.
Broncholithiasis is the presence of calcified substances in the trachea and bronchial tree. The main symptoms of broncholithiasis are cough, hemoptysis, obstructive pneumonia, and so forth. Broncholithiasis usually occurs in the right lung. It is common at the proximal end of the right middle bronchus and the bronchial opening at the anterior end of the right superior bronchus. Broncholithiasis is also accompanied by granulomatous diseases. In China, it is usually caused by tuberculosis infection. However, in European countries, it is correlated with histoplasmosis, Actinomyces infection, and tuberculosis.
The clinical symptoms of broncholithiasis are not specific. Cough and hemoptysis are common in the development of multiple diseases, and the incidence rate is low with <10 cases reported in children. The diagnosis of this disease is easy to be delayed and prone to be missed because of the lack of knowledge about this disease among medical staff. Chest CT of broncholithiasis can assist the diagnosis, but the final diagnosis depends on the discovery of calcified substances in the airway using bronchoscopy. During diagnosis and treatment of the patient in this study, the identification of stones using chest CT was not typical because the stones were inlaid in the granulation tissues. Bronchoscopy revealed incarceration of “bone-like substances” in the right superior lobe and the distal opening of the right middle bronchus. These substances had the characteristics of chipping off, easy dissolution, decalcification, and no tissue structure during preparation and staining, which were in line with the characteristics of stones. Furthermore, no bone manifestation occurred.
During broncholithiasis, usually mediastinal or hilar lymph nodes invade adjacent bronchial lumen through respiratory movement after tuberculosis and fungal infection. Broncholithiasis may also be caused by mucus in the diseased region, long-term inhalation of silicon dust and foreign bodies, and so forth. The calcified lymph nodes in pulmonary mediastinal histoplasmosis need several months or even years to progress into acute inflammation of granulation tissues and then to fibrosis. However, the time from calcified lymph nodes to broncholithiasis is unclear. Granulation tissue proliferation continuously occurred in the patient in this study during long-term stent implantation after trauma. Interventional bronchoscopy was performed multiple times, and no broncholithiasis occurred in the recent 4 years. Moreover, chest CT did not reveal calcification of mediastinal lymph nodes, and a local biopsy did not find lymph node structures. Pulmonary aspergillosis showed the same characteristics as stenosis and polyploid masses using bronchoscopy. Stone-like substances may be formed in some patients.[5,6] Jha et al reported that intrathecal aspergillosis combined with broncholithiasis was found in one case of allergic bronchopulmonary aspergillosis. However, such studies have not been reported in children. Pathologic examination suggested a large number of Aspergillus mycelia in the diseased region in the patient in this study who had no history of long-term inhalation of silicon dust and foreign bodies, which was consistent with previous findings. Therefore, broncholithiasis was considered to be a result of the fungal infection in the diseased region, but its underlying mechanism needs further investigation.
The basic aim of broncholithiasis treatment is to remove stones, release obstruction, and relieve clinical symptoms, which usually depends on endobronchial lithotomy or surgery. Bronchoscopy is superior because it can be used for not only diagnosis but also treatment. The patient in this study was treated using interventional bronchoscopy. A stent was implanted due to the damage of tracheal cartilage after trauma and luminal collapse, thereby improving local ventilation and secretion drainage. However, treatments against infection and polypnea were ineffective due to cough and polypnea for a long time. Multiple stones in granulation tissues in the diseased region found using bronchoscopy were removed with bronchoscopic laser treatment and clamping to avoid surgical trauma to the patient, thereby reducing the family burden to a large extent. Bronchoscopic lithotomy involved chances of stone fragments entering into the distal end of the bronchus, thus making the removal of these fragments difficult. However, massive hemorrhage was expected during lithotomy because large stones invaded the blood vessels. Surgical thoracotomy is usually used when bronchoscopic lithotomy fails or massive hemoptysis and tracheal fistula occur. However, it is associated with trauma. Lobectomy or segmentectomy should be performed if necessary. Hiroki Nishine reported that the stones on the bronchial wall could be moved by injecting saline into the bronchial lumen. Therefore, this method can be used in the future to reduce damage to the bronchial wall in patients with broncholithiasis. The culture of alveolar lavage fluid of the patient showed the growth of K pneumoniae and S aureus. Bronchoscopic specimen culture confirmed the presence of S aureus. The disease history showed that the patient had no fever. Bronchoscopic secretions reduced, related inflammatory indicators were basically normal, and the number of bacteria in the alveolar lavage fluid was low, which was considered to be Bacteriopexia. Therefore, anti-bacterial treatment was not performed. Previous studies indicated that antifungal agents should not be used when broncholithiasis was the only treatment for a pediatric patient with histoplasmosis because stones were usually formed after controlling fungal infection. However, the patient in this study had invasive pulmonary aspergillosis, and cough and polypnea did not occur after 1 month of oral administration of itraconazole. Moreover, bronchoscopy showed a few granulation tissues, but no broncholithiasis (Fig. 5C). Thus, the treatment was considered to be effective. Cough and polypnea did not occur in the patient, and antifungal treatment with itraconazole was continuously given for 1 month.
The complications of broncholithiasis include recurrent respiratory tract infection, bronchiectasis, tracheal fistula, and so forth. The primary complication of the patient in this study was a recurrent infection, which might be associated with luminal obstruction and inhibited secretion drainage due to stones. Massive hemoptysis might occur if pulmonary blood vessels were invaded by broncholithiasis. Therefore, timely diagnosis and treatment are of great importance.
In summary, although the incidence rate of broncholithiasis is low in children, the possibility of broncholithiasis should be considered when symptoms, such as recurrent cough, polypnea, hemoptysis, and respiratory tract infection, are difficult to be controlled, especially combined with tuberculosis and Aspergillus infection. Chest CT has a certain limitation in diagnosing broncholithiasis. Bronchoscopy should be improved as soon as possible to confirm the diagnosis, and stone removal should be performed using interventional bronchoscopy. A larger number of previously unknown diseases have gradually entered into the field of vision with the improvement in diagnosis and treatment. Multidisciplinary collaboration is important in the diagnosis and treatment of these diseases. The Departments of Respiratory Intervention, Radiology, and Pathology collaborated to understand and report the case of combined pulmonary aspergillosis and broncholithiasis while diagnosing and treating the patient in this study. However, the number of cases was low. Therefore, further clinical observation and summary are needed. Moreover, the mechanism underlying broncholithiasis caused by pulmonary aspergillosis should be further studied.
Conceptualization: Chen Meng.
Data curation: Chen Meng.
Formal analysis: Zhongxiao Zhang.
Investigation: Na Liu, Jing Ma.
Methodology: Xiuli Yan.
Project administration: Xia Liu.
Resources: Chen Meng, Zhongxiao Zhang, Na Liu.
Software: Xiuli Yan, Chong Shi, Xinxin Wang Coll.
Supervision: Chen Meng.
Validation: Zhongxiao Zhang, Xia Liu.
Visualization: Chong Shi, Xinxin Wang Coll.
Writing – original draft: Chen Meng, Jing Ma.
Writing – review & editing: Jing Ma.
. Jin YX, Jiang GN, Jiang L, et al. Diagnosis and treatment evaluation of 48 cases of broncholithiasis
. Thorac Cardiovasc Surg 2016;64:450–5.
. SB. Broncholithiasis
. Med J Chin People's Liberation Army 1989;2:149–50.
. Lee T, Woods C, O’Hagan A. Broncholithiasis
from histoplasmosis in a pediatric patient: case reports and review of literature. J Pediatric Infect Dis Soc 2013;2:76–9.
. Nishine H, Kurimoto N, Okamoto M, et al. Broncholithiasis
assessed by bronchoscopic saline solution injection. Intern Med 2015;54:1527–30.
. QL, JD, XZ. Performance of pulmonary aspergillosis
in bronchoscopy: analysis of 26 cases. Med Information 2011;24:5698–9.
. Chen D CW, Ji Ch. Diagnosis and treatment of pulmonary aspergillosis
by fiberoptic bronchoscopy: a 10 cases study. Chin J Tuberculosis Respiratory Dis 1997;20:316.
. Jha OK, Khanna A, Dabral C, et al. Tombs of Aspergillus: A missed cause of recurrent respiratory infections in allergic bronchopulmonary aspergillosis
. Indian J Crit Care Med 2016;20:421–4.
. Lim SY, Lee KJ, Jeon K, et al. Classification of broncholiths and clinical outcomes. Respirology 2013;18:637–42.
Keywords:Copyright © 2019 The Authors. Published by Wolters Kluwer Health, Inc. All rights reserved.
aspergillosis; broncholithiasis; children