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An unusual respiratory tract foreign body: A case report of pediatric broncholithiasis
International Journal of Pediatric Otorhinolaryngology Extra 13 (2016) 50–52
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology Extra j o u r n a l h o m e p a g e : w w w. e l s e v i e r. c o m / l o c a t e / i j p o r l
Case Report
An unusual respiratory tract foreign body: A case report of pediatric broncholithiasis Shuang Feng, Dabo Liu *, Zhenyun Huang, Jianwen Zhong, Jie Zhong Department of Otolaryngology, Guangzhou Women and Children’s Medical Center, Guangzhou 510120, China
A R T I C L E
I N F O
Article history: Received 18 February 2016 Received in revised form 3 May 2016 Accepted 5 May 2016 Available online 11 May 2016 Keywords: Broncholithiasis Foreign body Bronchoscopy
A B S T R A C T
Tracheobronchial foreign body aspiration is one of the most life-threatening diseases in children. Its symptom is non-specific sometimes; therefore the distinction with some similar clinical presentation pulmonary disease should be performed. Here, we reported a 5-year-old girl who was misdiagnosed with foreign body aspiration twice based on the broncho-obstructive symptom and radiologic finding. Finally, the diagnosis of broncholithiasis was confirmed by finding broncholiths in the respiratory tract through bronchoscopy. It is replied that bronchoscopy should be performed in the children with unexplained airway obstruction symptom. Furthermore, it is the first report about the broncholiths located at the upper main trachea. © 2016 Elsevier Ltd. All rights reserved.
1. Introduction Respiratory tract foreign body is a kind of common lifethreatening emergent disease in children. Foreign body aspiration history, classic symptoms, such as cough and wheezing, combined with thoracic CT are both helpful evidence for the diagnosis. However, some diseases, which have similar symptoms and radiological presentation, may be mistakenly considered as respiratory tract foreign body. Herein, we will report one case of broncholithiasis in a child, which had been considered as respiratory tract foreign body twice. 2. Case report A 5-year-old girl who presented with a cough that had persisted for >2 weeks and one episode of hemoptysis was admitted to the Department of Pulmonary Medicine in our hospital. The patient’s medical history revealed a cough, purulent sputum, recurrent fever, and hemoptysis for 2 weeks. The amount of hemoptysis was around 100 mL. The patient didn’t have the history of lithoptysis and the foreign body aspiration history; furthermore, she did not have a prior tuberculosis infection. This girl previously had a right thoracotomy because of a respiratory tract foreign body. After she was admitted to our hospital, her complete blood count were as follows: WBC, 28 × 109/L; N, 82%; HGB, 86 g/L; and PLT, 342 × 109/L. Fungal infection tests and tuberculosis antibody tests were normal.
* Corresponding author at: Dabo Liu, Department of Otolaryngology, Guangzhou Women and Children’s Medical Center, 318 Renmin Middle Road, Guangzhou 510120, China. Tel.: +86 02081330660; fax: 0086 020 38076020. E-mail address: [email protected] (D. Liu). http://dx.doi.org/10.1016/j.pedex.2016.05.001 1871-4048/© 2016 Elsevier Ltd. All rights reserved.
A chest radiograph revealed bilateral pulmonary infiltrations. She was diagnosed with pneumonia, and antibiotic therapy was immediately commenced. However, her cough, purulent sputum, and recurrent fevers continued. Thoracic CT revealed a dense shadow in the right main bronchus, accompanied by pulmonary infiltrations and bronchiectasis (Fig. 1A). No signs of calcification were observed in the chest. To explore additional details regarding the shadow in the bronchus, fiberoptic bronchoscopy was performed. It showed some brown stones that blocked the right main bronchus and appeared to be a foreign body surrounded by granulation hyperplasia (Fig. 2A). This patient was transferred to our department for rigid bronchoscopy. Eventually, several brown crystal objects were removed, the largest of which was 4 mm × 4 mm. The histological test of these objects showed that it belongs to calcified tissue. After the operation, the patient’s symptoms including her cough, purulent sputum, and fever gradually decreased. Improvement was also observed on a chest radiograph. She was discharged from the hospital after having antibiotic treatment for 2 days. We provided telephone follow-up for 6 months, and the patient’s abovementioned symptoms were relieved. However, she revisited our hospital 7 months later because of asthmatic symptoms lasting for 1 week. One week prior to that event, she was suddenly breathless while playing games. Her parents were sure that there was no history of foreign body aspiration. During this visit, she presented with a cough, asthma, and a recurrent fever, but no hemoptysis. A chest radiograph revealed a dense shadow that distorted the main trachea and was located about 2 cm beneath the glottis. It was again believed to be a respiratory tract foreign body, and rigid bronchoscopy was immediately performed for diagnostic and treatment purposes. A large calcified mass was found in and removed from the upper main trachea that was located about 2 cm beneath the entrance. It was proved to be the calcification
S. Feng et al. / International Journal of Pediatric Otorhinolaryngology Extra 13 (2016) 50–52
A
51
B
Fig. 1. Thoracic CT revealed a dense shadow in the right main bronchus (A). (B) shows the bronchus after the broncholiths’ removal in CT.
A
B
Fig. 2. A bronchoscopic image revealed a brown stone in the right main bronchus (A). (B) shows the bronchus after broncholith removal in bronchoscopic image.
substance by histological test post-operation. The diagnosis of broncholithiasis was confirmed. This broncholith was fragile and finally separated into pieces under pressing by the patient’s parent. The diameter of the largest one was 0.5 cm (Fig. 3). This girl’s symptoms were significantly relieved thereafter. She was discharged from the hospital thereafter. Four months later, thoracic CT and fiber bronchoscopy were again performed, and the results were normal (Figs. 1B and 2B). As of this report, based on data from telephone
Fig. 3. A photograph obtained during the second bronchoscopic broncholithectomy after broncholith removal. The broncholiths were fragile and broke into pieces after the operation.
follow-ups, the patient no longer has a cough, asthmatic symptoms, or a fever.
3. Discussion In our case, this patient was present with cough, breathless, and even hemoptysis. X-ray and CT both showed a dense shadow that blocked the respiratory tract. Moreover, fiberoptic bronchoscopy revealed something like stones in the bronchus. It seems that this case should be considered as tracheobronchial tract foreign body. To our knowledge, the history of foreign body aspiration is a critical evidence for the diagnosis of this kind of diseases. The origin of foreign body is mainly within two groups, food and objects. Nuts, beans, fruits and bones in the food group and toy or pen parts in the object group are the most common foreign bodies in children. However, in the present case, this patient did not have history of choking after inhalation of foreign body, especially in the second visit. The symptom happened suddenly according to her parents’ description. Therefore, these calcified stones carried out from respiratory tract should be endogenous. In our opinion, broncholithiasis may be the better diagnosis for this disease. To our knowledge, broncholithiasis is usually secondary to infection with Histoplasma capsulatum or Mycobacterium tuberculosis [1]. Actinomycosis, coccidioidomycosis, and cryptococcosis are also infectious causes [2]. The clinical presentation varies and includes a chronic cough, purulent sputum, hemoptysis, and episodic fevers. Lithoptysis is a representative symptom but is rarely reported. CT is useful for locating broncholiths in the bronchus. Distortion
52
S. Feng et al. / International Journal of Pediatric Otorhinolaryngology Extra 13 (2016) 50–52
secondary to a calcified endobronchial or peribronchial lymph node is a critical radiological finding [3]. Based on the location of a broncholith in the bronchus, there are three types of broncholiths: intraluminal, extraluminal, and mixed. Broncholiths can be visualized by bronchoscopy, which is helpful to provide key evidence for diagnosis. Therefore, chest CT combined with bronchoscopy has been considered as a reliable diagnostic method for broncholithiasis. Bronchoscopic broncholithectomy and thoracotomy are both treatment options for broncholith removal [4]. In case of extraluminal broncholiths, surgery is the only option. However, for loose broncholiths, bronchoscopic removal is a safe and effective option. It has been reported that 100% of intraluminal broncholiths and 67% of mixed broncholiths could be removed with bronchoscopy [5]. In our case, the calcified stone was found in the trachea and bronchus. Combined with the symptom and the result of the bronchoscopy, this should be a case of broncholithiasis in children. Broncholithiasis extremely rarely happens in children. In the past two decades, there were only three cases of pediatric broncholithiasis reported: one case with Mycobacterium kansasii infection, one case with ciliary dyskinesia, and the other one without mention etiology [5–8]. Though the clinical presentation is nonspecific in these cases, it is learned that there are some differences compared with the cases in adult. The formation of broncholiths in adult needs more than one year usually. However, it is rapid in children. In the case reported by Cuppen and colleagues, the broncholith was developed in only 8 months [6]. The time course in our case was even shorter, which was less than 3 weeks. In addition, it has been reported that most broncholiths were located in the right bronchial tree [9]. In this case, however, we found the broncholiths blocked in the upper main trachea, during the second bronchoscopic broncholithectomy. This is uncommon and has not been reported. Furthermore, in this patient, we did not find calcified sign in the chest CT. This is similar as that in the case reported by Cuppen et al. [6], which did not show any sign of broncholithiasis in either CT or mediastionscopy at first. It is replied that CT may be not as sensitive as bronchoscopy to locate the broncholith in early stage of broncholithiasis. Therefore, the clinical presentation of broncholithiasis, especially in children, is nonspecific and varies highly. It is likely to be misdiagnosed as other similar diseases, such as respiratory tract foreign body, pneumonia, or bronchiectasis only upon the symptom and radiologic finding. Bronchoscopy is a more reliable tool for diagnosis and treatment, as the broncholith could be discovered and removed directly using a bronchoscope.
To our knowledge, calcified lymph node, foreign body and bronchial cartilage plates are common sources of the broncholith. This patient did not have foreign body aspiration or infection of H. capsulatum or M. Tuberculosis. She also did not have any other pulmonary disease history, such as primary ciliary dyskinesia. Thus, the source of the broncholiths is still unclear in this case. One hypothesis is that the formation of broncholiths may be related with the previous thoracic surgery. The calcium in the residual blood may be deposited and gradually formed the nucleus of the broncholith after the operation. However, this hypothesis cannot explain the formation of the broncholith located at the upper main trachea. Hence, further study is needed to provide an insight into the broncholithiasis. We advocate that bronchoscopy should be performed in unexplained pulmonary diseases, especially in some children with airway obstruction and without history of foreign body aspiration. In conclusion, there is a high likelihood that broncholithiasis could be misdiagnosed in children and considered as a respiratory tract foreign body. CT might not be as important as bronchoscopy for the diagnosis of broncholithiasis at the early stage. Bronchoscopy is effective for diagnosis and therapy. It is the first time to report that the broncholiths could be located at the upper main trachea.
References [1] A.A. Ryberg, J.S. Gengler, V.A. Angelillo, W.J. Scott, Broncholithiasis: case report and literature review, Nebr. Med. J. 81 (1) (1996) 14–17. [2] A.S. Nollet, J.F. Vansteenkiste, M.G. Demedts, Broncholithiasis: rare but still present, Respir. Med. 92 (7) (1998) 963–965. [3] D.J. Conces Jr., R.D. Tarver, V.A. Vix, Broncholithiasis: CT features in 15 patients, AJR Am. J. Roentgenol. 157 (2) (1991) 249–253. [4] S.Y. Lim, K.J. Lee, K. Jeon, W.J. Koh, G.Y. Suh, M.P. Chung et al., Classification of broncholiths and clinical outcomes, Respirology 18 (4) (2013) 637–642. [5] E.J. Olson, J.P. Utz, U.B. Prakash, Therapeutic bronchoscopy in broncholithiasis, Am. J. Respir. Crit. Care Med. 160 (3) (1999) 766–770. [6] I. Cuppen, W.C. de Lange, S.S. de Graaf, S.J. Mol, C. Boetes, J.L. Yntema, Broncholithiasis in an immune compromised boy with disseminated Mycobacterium kansasii, Pediatr. Pulmonol. 42 (10) (2007) 980–983. [7] K. Hekimoglu, S. Gundogdu, Early adolescent primary ciliary dyskinesia associated with broncholithiasis, Pediatr. Pulmonol. 43 (7) (2008) 714–716. [8] T. Lee, C. Woods, A. O’Hagan, Broncholithiasis from histoplasmosis in a pediatric patient: case reports and review of literature, J. Pediatric. Infect. Dis. Soc. 2 (1) (2013) 76–79. [9] N.C. Hodgson, R.I. Inculet, Acute airway obstruction secondary to bilateral broncholithiasis, Chest 117 (4) (2000) 1205–1207.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology Extra j o u r n a l h o m e p a g e : w w w. e l s e v i e r. c o m / l o c a t e / i j p o r l
Case Report
An unusual respiratory tract foreign body: A case report of pediatric broncholithiasis Shuang Feng, Dabo Liu *, Zhenyun Huang, Jianwen Zhong, Jie Zhong Department of Otolaryngology, Guangzhou Women and Children’s Medical Center, Guangzhou 510120, China
A R T I C L E
I N F O
Article history: Received 18 February 2016 Received in revised form 3 May 2016 Accepted 5 May 2016 Available online 11 May 2016 Keywords: Broncholithiasis Foreign body Bronchoscopy
A B S T R A C T
Tracheobronchial foreign body aspiration is one of the most life-threatening diseases in children. Its symptom is non-specific sometimes; therefore the distinction with some similar clinical presentation pulmonary disease should be performed. Here, we reported a 5-year-old girl who was misdiagnosed with foreign body aspiration twice based on the broncho-obstructive symptom and radiologic finding. Finally, the diagnosis of broncholithiasis was confirmed by finding broncholiths in the respiratory tract through bronchoscopy. It is replied that bronchoscopy should be performed in the children with unexplained airway obstruction symptom. Furthermore, it is the first report about the broncholiths located at the upper main trachea. © 2016 Elsevier Ltd. All rights reserved.
1. Introduction Respiratory tract foreign body is a kind of common lifethreatening emergent disease in children. Foreign body aspiration history, classic symptoms, such as cough and wheezing, combined with thoracic CT are both helpful evidence for the diagnosis. However, some diseases, which have similar symptoms and radiological presentation, may be mistakenly considered as respiratory tract foreign body. Herein, we will report one case of broncholithiasis in a child, which had been considered as respiratory tract foreign body twice. 2. Case report A 5-year-old girl who presented with a cough that had persisted for >2 weeks and one episode of hemoptysis was admitted to the Department of Pulmonary Medicine in our hospital. The patient’s medical history revealed a cough, purulent sputum, recurrent fever, and hemoptysis for 2 weeks. The amount of hemoptysis was around 100 mL. The patient didn’t have the history of lithoptysis and the foreign body aspiration history; furthermore, she did not have a prior tuberculosis infection. This girl previously had a right thoracotomy because of a respiratory tract foreign body. After she was admitted to our hospital, her complete blood count were as follows: WBC, 28 × 109/L; N, 82%; HGB, 86 g/L; and PLT, 342 × 109/L. Fungal infection tests and tuberculosis antibody tests were normal.
* Corresponding author at: Dabo Liu, Department of Otolaryngology, Guangzhou Women and Children’s Medical Center, 318 Renmin Middle Road, Guangzhou 510120, China. Tel.: +86 02081330660; fax: 0086 020 38076020. E-mail address: [email protected] (D. Liu). http://dx.doi.org/10.1016/j.pedex.2016.05.001 1871-4048/© 2016 Elsevier Ltd. All rights reserved.
A chest radiograph revealed bilateral pulmonary infiltrations. She was diagnosed with pneumonia, and antibiotic therapy was immediately commenced. However, her cough, purulent sputum, and recurrent fevers continued. Thoracic CT revealed a dense shadow in the right main bronchus, accompanied by pulmonary infiltrations and bronchiectasis (Fig. 1A). No signs of calcification were observed in the chest. To explore additional details regarding the shadow in the bronchus, fiberoptic bronchoscopy was performed. It showed some brown stones that blocked the right main bronchus and appeared to be a foreign body surrounded by granulation hyperplasia (Fig. 2A). This patient was transferred to our department for rigid bronchoscopy. Eventually, several brown crystal objects were removed, the largest of which was 4 mm × 4 mm. The histological test of these objects showed that it belongs to calcified tissue. After the operation, the patient’s symptoms including her cough, purulent sputum, and fever gradually decreased. Improvement was also observed on a chest radiograph. She was discharged from the hospital after having antibiotic treatment for 2 days. We provided telephone follow-up for 6 months, and the patient’s abovementioned symptoms were relieved. However, she revisited our hospital 7 months later because of asthmatic symptoms lasting for 1 week. One week prior to that event, she was suddenly breathless while playing games. Her parents were sure that there was no history of foreign body aspiration. During this visit, she presented with a cough, asthma, and a recurrent fever, but no hemoptysis. A chest radiograph revealed a dense shadow that distorted the main trachea and was located about 2 cm beneath the glottis. It was again believed to be a respiratory tract foreign body, and rigid bronchoscopy was immediately performed for diagnostic and treatment purposes. A large calcified mass was found in and removed from the upper main trachea that was located about 2 cm beneath the entrance. It was proved to be the calcification
S. Feng et al. / International Journal of Pediatric Otorhinolaryngology Extra 13 (2016) 50–52
A
51
B
Fig. 1. Thoracic CT revealed a dense shadow in the right main bronchus (A). (B) shows the bronchus after the broncholiths’ removal in CT.
A
B
Fig. 2. A bronchoscopic image revealed a brown stone in the right main bronchus (A). (B) shows the bronchus after broncholith removal in bronchoscopic image.
substance by histological test post-operation. The diagnosis of broncholithiasis was confirmed. This broncholith was fragile and finally separated into pieces under pressing by the patient’s parent. The diameter of the largest one was 0.5 cm (Fig. 3). This girl’s symptoms were significantly relieved thereafter. She was discharged from the hospital thereafter. Four months later, thoracic CT and fiber bronchoscopy were again performed, and the results were normal (Figs. 1B and 2B). As of this report, based on data from telephone
Fig. 3. A photograph obtained during the second bronchoscopic broncholithectomy after broncholith removal. The broncholiths were fragile and broke into pieces after the operation.
follow-ups, the patient no longer has a cough, asthmatic symptoms, or a fever.
3. Discussion In our case, this patient was present with cough, breathless, and even hemoptysis. X-ray and CT both showed a dense shadow that blocked the respiratory tract. Moreover, fiberoptic bronchoscopy revealed something like stones in the bronchus. It seems that this case should be considered as tracheobronchial tract foreign body. To our knowledge, the history of foreign body aspiration is a critical evidence for the diagnosis of this kind of diseases. The origin of foreign body is mainly within two groups, food and objects. Nuts, beans, fruits and bones in the food group and toy or pen parts in the object group are the most common foreign bodies in children. However, in the present case, this patient did not have history of choking after inhalation of foreign body, especially in the second visit. The symptom happened suddenly according to her parents’ description. Therefore, these calcified stones carried out from respiratory tract should be endogenous. In our opinion, broncholithiasis may be the better diagnosis for this disease. To our knowledge, broncholithiasis is usually secondary to infection with Histoplasma capsulatum or Mycobacterium tuberculosis [1]. Actinomycosis, coccidioidomycosis, and cryptococcosis are also infectious causes [2]. The clinical presentation varies and includes a chronic cough, purulent sputum, hemoptysis, and episodic fevers. Lithoptysis is a representative symptom but is rarely reported. CT is useful for locating broncholiths in the bronchus. Distortion
52
S. Feng et al. / International Journal of Pediatric Otorhinolaryngology Extra 13 (2016) 50–52
secondary to a calcified endobronchial or peribronchial lymph node is a critical radiological finding [3]. Based on the location of a broncholith in the bronchus, there are three types of broncholiths: intraluminal, extraluminal, and mixed. Broncholiths can be visualized by bronchoscopy, which is helpful to provide key evidence for diagnosis. Therefore, chest CT combined with bronchoscopy has been considered as a reliable diagnostic method for broncholithiasis. Bronchoscopic broncholithectomy and thoracotomy are both treatment options for broncholith removal [4]. In case of extraluminal broncholiths, surgery is the only option. However, for loose broncholiths, bronchoscopic removal is a safe and effective option. It has been reported that 100% of intraluminal broncholiths and 67% of mixed broncholiths could be removed with bronchoscopy [5]. In our case, the calcified stone was found in the trachea and bronchus. Combined with the symptom and the result of the bronchoscopy, this should be a case of broncholithiasis in children. Broncholithiasis extremely rarely happens in children. In the past two decades, there were only three cases of pediatric broncholithiasis reported: one case with Mycobacterium kansasii infection, one case with ciliary dyskinesia, and the other one without mention etiology [5–8]. Though the clinical presentation is nonspecific in these cases, it is learned that there are some differences compared with the cases in adult. The formation of broncholiths in adult needs more than one year usually. However, it is rapid in children. In the case reported by Cuppen and colleagues, the broncholith was developed in only 8 months [6]. The time course in our case was even shorter, which was less than 3 weeks. In addition, it has been reported that most broncholiths were located in the right bronchial tree [9]. In this case, however, we found the broncholiths blocked in the upper main trachea, during the second bronchoscopic broncholithectomy. This is uncommon and has not been reported. Furthermore, in this patient, we did not find calcified sign in the chest CT. This is similar as that in the case reported by Cuppen et al. [6], which did not show any sign of broncholithiasis in either CT or mediastionscopy at first. It is replied that CT may be not as sensitive as bronchoscopy to locate the broncholith in early stage of broncholithiasis. Therefore, the clinical presentation of broncholithiasis, especially in children, is nonspecific and varies highly. It is likely to be misdiagnosed as other similar diseases, such as respiratory tract foreign body, pneumonia, or bronchiectasis only upon the symptom and radiologic finding. Bronchoscopy is a more reliable tool for diagnosis and treatment, as the broncholith could be discovered and removed directly using a bronchoscope.
To our knowledge, calcified lymph node, foreign body and bronchial cartilage plates are common sources of the broncholith. This patient did not have foreign body aspiration or infection of H. capsulatum or M. Tuberculosis. She also did not have any other pulmonary disease history, such as primary ciliary dyskinesia. Thus, the source of the broncholiths is still unclear in this case. One hypothesis is that the formation of broncholiths may be related with the previous thoracic surgery. The calcium in the residual blood may be deposited and gradually formed the nucleus of the broncholith after the operation. However, this hypothesis cannot explain the formation of the broncholith located at the upper main trachea. Hence, further study is needed to provide an insight into the broncholithiasis. We advocate that bronchoscopy should be performed in unexplained pulmonary diseases, especially in some children with airway obstruction and without history of foreign body aspiration. In conclusion, there is a high likelihood that broncholithiasis could be misdiagnosed in children and considered as a respiratory tract foreign body. CT might not be as important as bronchoscopy for the diagnosis of broncholithiasis at the early stage. Bronchoscopy is effective for diagnosis and therapy. It is the first time to report that the broncholiths could be located at the upper main trachea.
References [1] A.A. Ryberg, J.S. Gengler, V.A. Angelillo, W.J. Scott, Broncholithiasis: case report and literature review, Nebr. Med. J. 81 (1) (1996) 14–17. [2] A.S. Nollet, J.F. Vansteenkiste, M.G. Demedts, Broncholithiasis: rare but still present, Respir. Med. 92 (7) (1998) 963–965. [3] D.J. Conces Jr., R.D. Tarver, V.A. Vix, Broncholithiasis: CT features in 15 patients, AJR Am. J. Roentgenol. 157 (2) (1991) 249–253. [4] S.Y. Lim, K.J. Lee, K. Jeon, W.J. Koh, G.Y. Suh, M.P. Chung et al., Classification of broncholiths and clinical outcomes, Respirology 18 (4) (2013) 637–642. [5] E.J. Olson, J.P. Utz, U.B. Prakash, Therapeutic bronchoscopy in broncholithiasis, Am. J. Respir. Crit. Care Med. 160 (3) (1999) 766–770. [6] I. Cuppen, W.C. de Lange, S.S. de Graaf, S.J. Mol, C. Boetes, J.L. Yntema, Broncholithiasis in an immune compromised boy with disseminated Mycobacterium kansasii, Pediatr. Pulmonol. 42 (10) (2007) 980–983. [7] K. Hekimoglu, S. Gundogdu, Early adolescent primary ciliary dyskinesia associated with broncholithiasis, Pediatr. Pulmonol. 43 (7) (2008) 714–716. [8] T. Lee, C. Woods, A. O’Hagan, Broncholithiasis from histoplasmosis in a pediatric patient: case reports and review of literature, J. Pediatric. Infect. Dis. Soc. 2 (1) (2013) 76–79. [9] N.C. Hodgson, R.I. Inculet, Acute airway obstruction secondary to bilateral broncholithiasis, Chest 117 (4) (2000) 1205–1207.