Tracheal Diverticulum: A Rare Cause of Hoarseness of the Voice Ikram Chaudhry, FRCS(CTh), Hadi Mutairi, MD, Ebrahim Hassan, MD, Mussarat Afzal, MD, and Imtiaz Khurshid, MD Division of Thoracic Surgery, Chest Medicine, Radiology, and Anesthesia, King Fahad Specialist Hospital, Dammam, Saudi Arabia
Tracheal diverticulum is a rare benign disease also known as paratracheal air cyst. Mostly these are asymptomatic and are discovered incidentally on radiologic examination of the chest. Common symptoms are cough, recurrent respiratory tract infections, and sometimes dysphagia. Herein we report a rare case of hoarseness of voice caused by tracheal diverticulum. Computed tomography scan of neck revealed a large tracheal diverticulum, probably compressing the recurrent laryngeal nerve as bronchoscopy showed impaired right vocal cord movement. Two weeks after diverticulectomy, the patient’s voice returned to normal. (Ann Thorac Surg 2014;97:e29–31) Ó 2014 by The Society of Thoracic Surgeons
T
racheal diverticulum, or paratracheal cyst, is an uncommonly encountered and reported clinical entity. Reported data are either single case reports or small case series. Tracheal diverticulum was first reported by Rokitnsky in 1938, as cited by Mathey and associates [1], when he made anatomicopathologic observation of 3 cases of tracheal diverticulum. Tracheal diverticulum is a relatively benign and most commonly asymptomatic entity. The exact prevalence of tracheal diverticulum is unknown. The incidence of tracheal diverticulum is approximately 1% in adults and 0.3% in children based on the findings of serial 800 autopsy reports by MacKinnon [2]. A 54-year-old nonsmoking woman presented to our clinic with a history of mild chronic cough and a 2-month history of hoarseness. She had no history of dyspnea on exertion, hemoptysis, or dysphagia. Physical examination of the neck and the chest was unremarkable. Computed tomography scan of the neck and chest with threedimensional reconstruction revealed a 3 4 cm tracheal diverticulum in the right posterolateral retrotracheal position at the level of the fourth and fifth tracheal ring (Figs 1A, 1B). Routine blood investigation and spirometric values were normal. Barium swallow study was normal (Fig 1C). Flexible bronchoscopy showed the right vocal cord movement was impaired, and there was a diverticulum in the upper trachea having a pinhole connection with the trachea. The diverticulum was approached
Accepted for publication Sept 23, 2013. Address correspondence to Dr Chaudhry, Division of Thoracic Surgery, King Fahad Specialist Hospital, PO Box 15215, Dammam SA 31444, Saudi Arabia; e-mail: [email protected].
Ó 2014 by The Society of Thoracic Surgeons Published by Elsevier Inc
through right lateral cervical incision. After careful dissection, the right recurrent laryngeal nerve was dissected free of the diverticulum. The diverticulum was excised using a surgical stapler (Fig 2A). The patient was extubated on the operating table and postoperative recovery was uneventful. Histopathology examination of the specimen showed a cystic mass lined with respiratory epithelium with no cartilages consistent with acquired tracheal diverticulum (Fig 2B). At 3-week follow-up, the wound had healed nicely and the patient’s voice had returned to normal. Follow-up flexible bronchoscopic examination revealed normal movement of the right vocal cord and no tracheal diverticulum, and the rest of the trachea was normal. A 3-month follow-up computed tomography scan of the neck was normal (Fig 1D).
Comment Tracheal diverticulum is also known as air cysts of tracheal origin. Cysts of tracheal origin are divided into three types—tracheogenic cysts, tracheocele, and tracheal diverticulum—based on the histology and size. Tracheogenic cyst wall contains smooth muscle and cartilage whereas the other two are composed of respiratory epithelium only. Based on their size, these are called tracheocele if less than 2 cm and diverticula if greater than 2 cm [3]. Tracheal diverticula are basically classified as congenital or acquired based on the anatomic location and histologic criteria. Congenital tracheal diverticula are usually small and commonly found 4 to 5 cm below the vocal cords or sometimes above the carina. Acquired tracheal diverticula are large and are commonly found in the posterolateral region between the intrathoracic and extrathoracic trachea; the wall is composed of respiratory epithelium and devoid of smooth muscle and cartilages, as is in our case [4]. Katz and associates [5] have grouped diverticula into four types: rudimentary bronchus, cystic dilation of mucous gland duct, tracheocele, and diverticulum with tracheobronchomegaly. Goo and colleagues [6] reported a series of 64 cases of tracheal diverticulum in living humans. In all these patients, there was obstructed pattern in pulmonary function studies. Acquired tracheal diverticulum is a result of increased intraluminal pressure due to chronic cough as in chronic obstructive airway disease or emphysema and in professions that require excessive vocal cord or pulmonary efforts, leading to herniation of membranous part of the trachea [6]. Commonly they occur in the right posterolateral position, which is unprotected space as compared with the left, which is protected by the arch of aorta and esophagus. They act as a reservoir for secretions. Tracheal diverticulum is a rare clinical entity, however. Patients can present with symptoms of recurrent respiratory tract infections, cough, dyspnea, hemoptysis, painful neck swelling, cervical abscess, globuspharygeus, respiratory distress, and sometimes with dysphagia [7]. The differential diagnosis of tracheal diverticulum includes pharyngocele, laryngocele, Zenker’s diverticulum, 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.09.069
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CASE REPORT CHAUDHRY ET AL HOARSENESS CAUSED BY TRACHEAL DIVERTICULUM
Ann Thorac Surg 2014;97:e29–31
Fig 1. (A) Computed tomography scan of the neck showing an air-containing cyst adjacent to the right tracheal wall (white arrow). (B) Reconstructed image of the airway showing the tracheal diverticulum at the thoracic inlet (white arrow). (C) Normal esophagogram. (D) Computed tomography scan of the neck after surgery.
apical lung herniation, and apical paraspetal blebs or bullae. Computed tomography with three-dimensional reconstruction is the best imaging modality for diagnosis and planning surgery. Barium examination is very useful for identifying laryngocele, pharyngocele, and Zenker’s diverticulum. Fig 2. (A) The resected specimen. (B) The microscopic examination showing a ciliated simple respiratory epithelium (hematoxylin & eosin stain, magnification 10).
In most cases, tracheal diverticulum can be treated conservatively with antibiotics and mucolytic agents. In symptomatic patients, there are different options for treatment, including endoscopic laser cauterization, electrocoagulation, and surgical resection. Tracheal intubation sometimes can be very difficult in such cases, and
Ann Thorac Surg 2014;97:e29–31
respiratory distress and pneumomediastinum due to selective intubation and perforation of the diverticulum has been reported [8]. Hoarseness of voice due to tracheal diverticulum is an extremely rare but curable disease, as in our patient; after diverticulectomy, her voice returned to normal. To the best of our knowledge, this is the first case reported in the English medical literature in which hoarseness was caused by a tracheal diverticulum. So, when a patient presents with hoarseness, tracheal diverticulum can be a possible cause and should be ruled out.
References 1. Mathey S, Assumes IK, Rosales JK. Congenital tracheal diverticulum and tracheo-oesophageal fistula without esophageal atresia. Thorax 1954;9:106–11.
CASE REPORT CHAUDHRY ET AL HOARSENESS CAUSED BY TRACHEAL DIVERTICULUM
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2. MacKinnon D. Tracheal diverticulum. J Path Bacteriol 1953;65: 513–7. 3. Grassy R, Rea G, Scagillone M, Brunese L, Scilapi M. Imaging of tracheocele: report of three cases and review of literature. Radiol Med (Torino) 2000;100: 285–7. 4. Early EK, Bothwell MR. Congenital tracheal diverticulum. Otolaryngol Head Neck Surg 2002;127:119–21. 5. Katz I, Levine M, Herman P. Tracheobronchomegaly. AJR Am J Roentgenol 1962;88:1084–9. 6. Goo JM, Im J, Ahn KM, et al. Right paratracheal air cysts in the thoracic inlet: clinical and radiological significance. AJR Am J Roentgenol 1999;173: 65–70. 7. Enrique Javier Soto-Hurtado E, Penuela-Ruiz L, RiveraSanches I, Torres-Timenez J. Tracheal diverticulum: a review of literature. Lung 2006;184:303–7. 8. Moller GM, Ten Berge EJ, Stassen CM. Tracheocele a rare cause of difficult intubation and subsequent pneumomediastinum. Eur Respir J 1994;7:1376–7.
Tracheal diverticulum is a rare benign disease also known as paratracheal air cyst. Mostly these are asymptomatic and are discovered incidentally on radiologic examination of the chest. Common symptoms are cough, recurrent respiratory tract infections, and sometimes dysphagia. Herein we report a rare case of hoarseness of voice caused by tracheal diverticulum. Computed tomography scan of neck revealed a large tracheal diverticulum, probably compressing the recurrent laryngeal nerve as bronchoscopy showed impaired right vocal cord movement. Two weeks after diverticulectomy, the patient’s voice returned to normal. (Ann Thorac Surg 2014;97:e29–31) Ó 2014 by The Society of Thoracic Surgeons
T
racheal diverticulum, or paratracheal cyst, is an uncommonly encountered and reported clinical entity. Reported data are either single case reports or small case series. Tracheal diverticulum was first reported by Rokitnsky in 1938, as cited by Mathey and associates [1], when he made anatomicopathologic observation of 3 cases of tracheal diverticulum. Tracheal diverticulum is a relatively benign and most commonly asymptomatic entity. The exact prevalence of tracheal diverticulum is unknown. The incidence of tracheal diverticulum is approximately 1% in adults and 0.3% in children based on the findings of serial 800 autopsy reports by MacKinnon [2]. A 54-year-old nonsmoking woman presented to our clinic with a history of mild chronic cough and a 2-month history of hoarseness. She had no history of dyspnea on exertion, hemoptysis, or dysphagia. Physical examination of the neck and the chest was unremarkable. Computed tomography scan of the neck and chest with threedimensional reconstruction revealed a 3 4 cm tracheal diverticulum in the right posterolateral retrotracheal position at the level of the fourth and fifth tracheal ring (Figs 1A, 1B). Routine blood investigation and spirometric values were normal. Barium swallow study was normal (Fig 1C). Flexible bronchoscopy showed the right vocal cord movement was impaired, and there was a diverticulum in the upper trachea having a pinhole connection with the trachea. The diverticulum was approached
Accepted for publication Sept 23, 2013. Address correspondence to Dr Chaudhry, Division of Thoracic Surgery, King Fahad Specialist Hospital, PO Box 15215, Dammam SA 31444, Saudi Arabia; e-mail: [email protected].
Ó 2014 by The Society of Thoracic Surgeons Published by Elsevier Inc
through right lateral cervical incision. After careful dissection, the right recurrent laryngeal nerve was dissected free of the diverticulum. The diverticulum was excised using a surgical stapler (Fig 2A). The patient was extubated on the operating table and postoperative recovery was uneventful. Histopathology examination of the specimen showed a cystic mass lined with respiratory epithelium with no cartilages consistent with acquired tracheal diverticulum (Fig 2B). At 3-week follow-up, the wound had healed nicely and the patient’s voice had returned to normal. Follow-up flexible bronchoscopic examination revealed normal movement of the right vocal cord and no tracheal diverticulum, and the rest of the trachea was normal. A 3-month follow-up computed tomography scan of the neck was normal (Fig 1D).
Comment Tracheal diverticulum is also known as air cysts of tracheal origin. Cysts of tracheal origin are divided into three types—tracheogenic cysts, tracheocele, and tracheal diverticulum—based on the histology and size. Tracheogenic cyst wall contains smooth muscle and cartilage whereas the other two are composed of respiratory epithelium only. Based on their size, these are called tracheocele if less than 2 cm and diverticula if greater than 2 cm [3]. Tracheal diverticula are basically classified as congenital or acquired based on the anatomic location and histologic criteria. Congenital tracheal diverticula are usually small and commonly found 4 to 5 cm below the vocal cords or sometimes above the carina. Acquired tracheal diverticula are large and are commonly found in the posterolateral region between the intrathoracic and extrathoracic trachea; the wall is composed of respiratory epithelium and devoid of smooth muscle and cartilages, as is in our case [4]. Katz and associates [5] have grouped diverticula into four types: rudimentary bronchus, cystic dilation of mucous gland duct, tracheocele, and diverticulum with tracheobronchomegaly. Goo and colleagues [6] reported a series of 64 cases of tracheal diverticulum in living humans. In all these patients, there was obstructed pattern in pulmonary function studies. Acquired tracheal diverticulum is a result of increased intraluminal pressure due to chronic cough as in chronic obstructive airway disease or emphysema and in professions that require excessive vocal cord or pulmonary efforts, leading to herniation of membranous part of the trachea [6]. Commonly they occur in the right posterolateral position, which is unprotected space as compared with the left, which is protected by the arch of aorta and esophagus. They act as a reservoir for secretions. Tracheal diverticulum is a rare clinical entity, however. Patients can present with symptoms of recurrent respiratory tract infections, cough, dyspnea, hemoptysis, painful neck swelling, cervical abscess, globuspharygeus, respiratory distress, and sometimes with dysphagia [7]. The differential diagnosis of tracheal diverticulum includes pharyngocele, laryngocele, Zenker’s diverticulum, 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2013.09.069
e30
CASE REPORT CHAUDHRY ET AL HOARSENESS CAUSED BY TRACHEAL DIVERTICULUM
Ann Thorac Surg 2014;97:e29–31
Fig 1. (A) Computed tomography scan of the neck showing an air-containing cyst adjacent to the right tracheal wall (white arrow). (B) Reconstructed image of the airway showing the tracheal diverticulum at the thoracic inlet (white arrow). (C) Normal esophagogram. (D) Computed tomography scan of the neck after surgery.
apical lung herniation, and apical paraspetal blebs or bullae. Computed tomography with three-dimensional reconstruction is the best imaging modality for diagnosis and planning surgery. Barium examination is very useful for identifying laryngocele, pharyngocele, and Zenker’s diverticulum. Fig 2. (A) The resected specimen. (B) The microscopic examination showing a ciliated simple respiratory epithelium (hematoxylin & eosin stain, magnification 10).
In most cases, tracheal diverticulum can be treated conservatively with antibiotics and mucolytic agents. In symptomatic patients, there are different options for treatment, including endoscopic laser cauterization, electrocoagulation, and surgical resection. Tracheal intubation sometimes can be very difficult in such cases, and
Ann Thorac Surg 2014;97:e29–31
respiratory distress and pneumomediastinum due to selective intubation and perforation of the diverticulum has been reported [8]. Hoarseness of voice due to tracheal diverticulum is an extremely rare but curable disease, as in our patient; after diverticulectomy, her voice returned to normal. To the best of our knowledge, this is the first case reported in the English medical literature in which hoarseness was caused by a tracheal diverticulum. So, when a patient presents with hoarseness, tracheal diverticulum can be a possible cause and should be ruled out.
References 1. Mathey S, Assumes IK, Rosales JK. Congenital tracheal diverticulum and tracheo-oesophageal fistula without esophageal atresia. Thorax 1954;9:106–11.
CASE REPORT CHAUDHRY ET AL HOARSENESS CAUSED BY TRACHEAL DIVERTICULUM
e31
2. MacKinnon D. Tracheal diverticulum. J Path Bacteriol 1953;65: 513–7. 3. Grassy R, Rea G, Scagillone M, Brunese L, Scilapi M. Imaging of tracheocele: report of three cases and review of literature. Radiol Med (Torino) 2000;100: 285–7. 4. Early EK, Bothwell MR. Congenital tracheal diverticulum. Otolaryngol Head Neck Surg 2002;127:119–21. 5. Katz I, Levine M, Herman P. Tracheobronchomegaly. AJR Am J Roentgenol 1962;88:1084–9. 6. Goo JM, Im J, Ahn KM, et al. Right paratracheal air cysts in the thoracic inlet: clinical and radiological significance. AJR Am J Roentgenol 1999;173: 65–70. 7. Enrique Javier Soto-Hurtado E, Penuela-Ruiz L, RiveraSanches I, Torres-Timenez J. Tracheal diverticulum: a review of literature. Lung 2006;184:303–7. 8. Moller GM, Ten Berge EJ, Stassen CM. Tracheocele a rare cause of difficult intubation and subsequent pneumomediastinum. Eur Respir J 1994;7:1376–7.
