Case Study
Mediastinal bronchogenic cyst compressing left recurrent laryngeal nerve
Asian Cardiovascular & Thoracic Annals 21(6) 729–731 ß The Author(s) 2012 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492312467542 aan.sagepub.com
Hakeem Zubair Ashraf, Abdul Ghani Ahangar, Ghulam Nabi Lone, Shyam Singh Rathore and Masrat Ashai
Abstract A 15-year-old boy developed recurrent laryngeal nerve compression by a bronchogenic cyst, causing recurrent choking. Bronchogenic cyst compressing the recurrent laryngeal nerve is unusual. The patient had a normal voice and occasional choking with periods of normalcy in between. A left posterolateral thoracotomy was performed and cyst was excised. Histopathology revealed bronchogenic cyst.
Keywords Airway obstruction, bronchogenic cyst, recurrent laryngeal nerve
Case report A 15-year-old boy was referred to us with a history of recurrent chest infections and episodes of recurrent choking in the previous 3 years. He also reported history of occasional vague mild retrosternal pain. There was no history of hoarseness. He had seen an ear, nose, and throat surgeon on number of occasions, and
physical examinations and laryngoscopy were reported as normal. An acute upper respiratory tract infection took him to a general physician who ordered a chest radiograph that revealed a cystic mass in the left hilar region (Figure 1). A provisional diagnosis of mediastinal hydatid cyst was made, but the possibilities of aortic aneurysm and bronchogenic cyst were also kept in mind. A subsequent chest computed tomography scan revealed a cystic mass measuring 3 5 cm in the mediastinum, abutting the left main pulmonary artery and aorta but not communicating with them (Figure 2). A left posterolateral thoracotomy was performed. After retracting the lung medially, the cyst was exposed and found in the aortopulmonary window, abutting the pulmonary artery and aorta, and compressing the recurrent laryngeal nerve (RLN) below the aortic arch (Figure 3). The cyst was shaved off from the pulmonary artery and aorta, and seen as pedicled with the left RLN (Figure 4). It was not communicating with any of the great vessels or bronchi. The nerve was attached to the cyst by strands of fibrous tissue. Incision of the
Department of CVTS, SKIMS, Soura, Srinagar, Kashmir, India
Figure 1. Chest radiograph showing a left cystic mass.
Corresponding author: Hakeem Zubair Ashraf, Department of CVTS, SKIMS, Soura, Srinagar, Jammu & Kashmir, 190001 India. Email: [email protected]
730
Asian Cardiovascular & Thoracic Annals 21(6)
Figure 2. Computed tomography scan showing a posterior hilar cystic shadow.
Figure 3. Preoperative photograph showing attachment of the cyst.
cyst revealed mucoid viscous fluid, dirty white in color. Histopathology confirmed bronchogenic cyst. The patient was relieved of his symptoms one year after his surgery.
Discussion Bronchogenic cysts are relatively rare yet they represent the most common cystic lesion of the mediastinum. The location of bronchogenic cysts is variable and they can
be found along the tracheoesophageal course, in perihilar or intraparenchymal sites, with a predilection for the area around the carina. Those in the mediastinum frequently attach to, but do not communicate with, the tracheobronchial tree. More than 50% of cases are diagnosed in patients older than 15 years. Reported prevalence rates are 1 case per 42,000 or 1 case per 68,000 of the population.1 Although asymptomatic bronchogenic cysts are known and are incidentally found on radiography, most cysts are symptomatic.
Ashraf et al.
731 In our patient, the mediastinal bronchogenic cyst was compressing the left RLN, causing repeated episodes of choking and vague retrosternal pain. The recurrent episodes of choking may have been due to recurrent transient neurapraxia of the left RLN, leading to mild aspiration. Despite chronic compression of the left RLN by the cyst, there was no palsy of the left vocal cord observed, which was unusual. Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
Conflicts of interest statement None declared.
References Figure 4. The cyst adhering to the recurrent laryngeal nerve by fibrous strands.
Adults commonly present with chest pain and dysphagia, whereas children and infants most often present with symptoms of airway or esophageal compression. Superior vena cava syndrome, tracheal compression, pneumothorax, pleurisy, and pneumonia have also been reported.2 RLN compression by bronchogenic cyst is rare. Takahashi and colleagues3 demonstrated bilateral RLN palsy due to an esophageal bronchogenic cyst. Rice and Putnam4 reported left-sided RLN palsy caused by a recurrent mediastinal bronchogenic cyst. Koga and colleagues5 reported thoracoscopic excision of a bronchogenic cyst causing RLN palsy.
1. Limaı¨ em F, Ayadi KA, Djilani H, Kilani T and El Mezni F. Pulmonary and mediastinal bronchogenic cysts: a clinicopathologic study of 33 cases. Lung 2008; 186: 55–61. 2. Aktogu S, Yuncu G, Halilc¸olar H, Ermete S and Buduneli T. Bronchogenic cysts: Clinicopathological presentation and treatment. Eur Respir J 1996; 9: 2017–2021. 3. Takahashi Y, Ishikawa S, Ikegami N, Tamada J and Matsukura T. Case of bronchogenic cyst in the esophageal wall. Nippon Kyobu Geka Gakkai Zasshi 1994; 42: 1944–1948. 4. Rice DC and Putnam JB Jr. Recurrent bronchogenic cyst causing recurrent laryngeal nerve palsy. Eur J Cardiothorac Surg 2002; 21: 561–563. 5. Koga Y, Uchiyama A, Noshiro H, et al. Complete extirpation of a bronchogenic cyst causing recurrent laryngeal nerve palsy by thoracoscopy: report of a case. Surg Today 2006; 36: 79–81.
Mediastinal bronchogenic cyst compressing left recurrent laryngeal nerve
Asian Cardiovascular & Thoracic Annals 21(6) 729–731 ß The Author(s) 2012 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492312467542 aan.sagepub.com
Hakeem Zubair Ashraf, Abdul Ghani Ahangar, Ghulam Nabi Lone, Shyam Singh Rathore and Masrat Ashai
Abstract A 15-year-old boy developed recurrent laryngeal nerve compression by a bronchogenic cyst, causing recurrent choking. Bronchogenic cyst compressing the recurrent laryngeal nerve is unusual. The patient had a normal voice and occasional choking with periods of normalcy in between. A left posterolateral thoracotomy was performed and cyst was excised. Histopathology revealed bronchogenic cyst.
Keywords Airway obstruction, bronchogenic cyst, recurrent laryngeal nerve
Case report A 15-year-old boy was referred to us with a history of recurrent chest infections and episodes of recurrent choking in the previous 3 years. He also reported history of occasional vague mild retrosternal pain. There was no history of hoarseness. He had seen an ear, nose, and throat surgeon on number of occasions, and
physical examinations and laryngoscopy were reported as normal. An acute upper respiratory tract infection took him to a general physician who ordered a chest radiograph that revealed a cystic mass in the left hilar region (Figure 1). A provisional diagnosis of mediastinal hydatid cyst was made, but the possibilities of aortic aneurysm and bronchogenic cyst were also kept in mind. A subsequent chest computed tomography scan revealed a cystic mass measuring 3 5 cm in the mediastinum, abutting the left main pulmonary artery and aorta but not communicating with them (Figure 2). A left posterolateral thoracotomy was performed. After retracting the lung medially, the cyst was exposed and found in the aortopulmonary window, abutting the pulmonary artery and aorta, and compressing the recurrent laryngeal nerve (RLN) below the aortic arch (Figure 3). The cyst was shaved off from the pulmonary artery and aorta, and seen as pedicled with the left RLN (Figure 4). It was not communicating with any of the great vessels or bronchi. The nerve was attached to the cyst by strands of fibrous tissue. Incision of the
Department of CVTS, SKIMS, Soura, Srinagar, Kashmir, India
Figure 1. Chest radiograph showing a left cystic mass.
Corresponding author: Hakeem Zubair Ashraf, Department of CVTS, SKIMS, Soura, Srinagar, Jammu & Kashmir, 190001 India. Email: [email protected]
730
Asian Cardiovascular & Thoracic Annals 21(6)
Figure 2. Computed tomography scan showing a posterior hilar cystic shadow.
Figure 3. Preoperative photograph showing attachment of the cyst.
cyst revealed mucoid viscous fluid, dirty white in color. Histopathology confirmed bronchogenic cyst. The patient was relieved of his symptoms one year after his surgery.
Discussion Bronchogenic cysts are relatively rare yet they represent the most common cystic lesion of the mediastinum. The location of bronchogenic cysts is variable and they can
be found along the tracheoesophageal course, in perihilar or intraparenchymal sites, with a predilection for the area around the carina. Those in the mediastinum frequently attach to, but do not communicate with, the tracheobronchial tree. More than 50% of cases are diagnosed in patients older than 15 years. Reported prevalence rates are 1 case per 42,000 or 1 case per 68,000 of the population.1 Although asymptomatic bronchogenic cysts are known and are incidentally found on radiography, most cysts are symptomatic.
Ashraf et al.
731 In our patient, the mediastinal bronchogenic cyst was compressing the left RLN, causing repeated episodes of choking and vague retrosternal pain. The recurrent episodes of choking may have been due to recurrent transient neurapraxia of the left RLN, leading to mild aspiration. Despite chronic compression of the left RLN by the cyst, there was no palsy of the left vocal cord observed, which was unusual. Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
Conflicts of interest statement None declared.
References Figure 4. The cyst adhering to the recurrent laryngeal nerve by fibrous strands.
Adults commonly present with chest pain and dysphagia, whereas children and infants most often present with symptoms of airway or esophageal compression. Superior vena cava syndrome, tracheal compression, pneumothorax, pleurisy, and pneumonia have also been reported.2 RLN compression by bronchogenic cyst is rare. Takahashi and colleagues3 demonstrated bilateral RLN palsy due to an esophageal bronchogenic cyst. Rice and Putnam4 reported left-sided RLN palsy caused by a recurrent mediastinal bronchogenic cyst. Koga and colleagues5 reported thoracoscopic excision of a bronchogenic cyst causing RLN palsy.
1. Limaı¨ em F, Ayadi KA, Djilani H, Kilani T and El Mezni F. Pulmonary and mediastinal bronchogenic cysts: a clinicopathologic study of 33 cases. Lung 2008; 186: 55–61. 2. Aktogu S, Yuncu G, Halilc¸olar H, Ermete S and Buduneli T. Bronchogenic cysts: Clinicopathological presentation and treatment. Eur Respir J 1996; 9: 2017–2021. 3. Takahashi Y, Ishikawa S, Ikegami N, Tamada J and Matsukura T. Case of bronchogenic cyst in the esophageal wall. Nippon Kyobu Geka Gakkai Zasshi 1994; 42: 1944–1948. 4. Rice DC and Putnam JB Jr. Recurrent bronchogenic cyst causing recurrent laryngeal nerve palsy. Eur J Cardiothorac Surg 2002; 21: 561–563. 5. Koga Y, Uchiyama A, Noshiro H, et al. Complete extirpation of a bronchogenic cyst causing recurrent laryngeal nerve palsy by thoracoscopy: report of a case. Surg Today 2006; 36: 79–81.
