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Chamberlain Mediastinotomy for Diagnosis of Adenoid Cystic Carcinoma of Trachea
Case Reports
515 CASE REPORTS
Heart, Lung and Circulation 2008;17:505–518
Chamberlain Mediastinotomy for Diagnosis of Adenoid Cystic Carcinoma of Trachea Saulat H. Fatimi, MD, FACS a , Rubina A. Sajwani, MBBS a , Nadeem Rizvi, MBBS b and Muhammad A. Javed, MD a Asmatullah Khan, MBBS a Usman Ahmad, MD a,∗ a
Department of Surgery, Division of Cardiothoracic Surgery, Aga Khan University, Karachi, Pakistan b Division of Chest Diseases, Jinnah Post Graduate Medical Center, Karachi, Pakistan
Primary tracheal tumours are extremely rare. Bronchoscopy is the standard diagnostic procedure of obtaining biopsy of a tracheal mass, however it becomes challenging if the obstructing lesion is placed distally along the trachea occluding greater than 90% of the airway. We report the case of a 25-year-old male who suffered from severe tracheal stenosis. The lesion was biopsied through a chamberlain mediastinotomy, under local and mask anaesthesia and was found to be primary adenoid cystic carcinoma. (Heart, Lung and Circulation 2008;17:505–518) © 2007 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved. Keywords. Chamberlain’s mediastinotomy; Anterior mediastinotomy; Adenoid cystic carcinoma; Trachea
Introduction
P
rimary tracheal malignancy has an incidence of <2 million persons per year.1 Malignant lesions rarely cause tracheal stenosis, but when present, adenoid cystic carcinoma (ACC) is the second most common histologic type.1,2 Rigid bronchoscopy under general anaesthesia (GA) is the conventional technique employed to biopsy,1–3 but management of airway in patients with severe tracheal obstruction is challenging for both the surgeon and the anaesthesiologist.4 We report our successful experience of establishing diagnosis of primary tracheal ACC in a patient with severe tracheal stenosis using chamberlain’s mediastinotomy under local and mask anaesthesia.
Case Presentation A 25-year-old nonsmoker male presented with progressively worsening dyspnoea for 2 years and exertional dyspnoea for 5 months. He was initially treated for asthma at another hospital but his condition soon deteriorated and he developed severe breathlessness on lying supine. Chest roentgenogram and thoracic computed tomographic scan showed a homogenous circumtracheal mass extending from lower part of trachea to the carina obstructing greater Received 11 February 2007; received in revised form 21 July 2007; accepted 30 July 2007; available online 10 October 2007 ∗ Corresponding author at: Department of Surgery, Yale University, Boyer Center for Molecular Medicine, Room 437, 295 Congress Ave, New Haven, CT 06510, USA. Tel.: +1 203 737 2289. E-mail address: [email protected] (U. Ahmad).
than 85% of the tracheal lumen. He was referred to our centre for an elective biopsy. The patient had no past history of tuberculosis or other medical comorbids including diabetes, hypertension, asthma, gastro-oesophageal reflux or allergy. Chest examination revealed grade IV wheezing and expiratory stridor. Chest wall retractions were also observed. Arterial blood gases in room air showed mild hypoxaemia and CO2 retention, with PaO2 = 75 mmHg, PaCO2 = 46 mmHg and oxygen saturation of 96%. Considering the patients’ inability to lie supine and to undergo intubation, Chamberlain’s mediastinotomy was carried out under local and inhalation anaesthesia, with the patient in a semi-recumbent position. After infiltrating the area with 2% xylocaine and epinephrine, an incision was made above the third rib in the second intercostal space on the right side. The superior vena cava, phrenic nerve, aorta and trachea were identified. The trachea was narrow and fibrotic. Multiple biopsy samples of tissue around the trachea and carina along with several paratracheal lymph nodes were taken. Frozen section was performed to ensure that the tissue obtained was a representative specimen. Histopathological examination showed the biopsy specimen to be positive for epithelial carcinoma. Final histopathological examination confirmed infiltrating ACC (Fig. 1). Post-operatively, the patient required 3 days to recover and had to be observed for a day as he was retaining CO2 . There were no major operative complications. Unfortunately further management was hindered as the patient failed to follow up.
© 2007 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved.
1443-9506/04/$30.00 doi:10.1016/j.hlc.2007.07.008
516
Case Reports
CASE REPORTS
Heart, Lung and Circulation 2008;17:505–518
Fig. 1. Adenoid cystic carcinoma: H&E stain. (A) 20× and (B) 40×.
Discussion Neoplastic lesions are an extremely rare cause of tracheal stenosis.1 ACC is the second most common primary tracheal malignancy,1,2 and is characterised by slow growth and infiltrating nature often leading to late diagnosis. However it is frequently amenable to surgical resection with a good long term survival.1,2 The typical presenting symptoms are those of airway obstruction, i.e. wheezing, progressive dyspnoea, stridor, cough and haemoptysis so it is not uncommon for these symptoms to be mistaken for asthma.1,2 Chest X-ray in these patients is often normal,1,3 therefore CT scan (95% sensitive) is highly recommended to provide early detection of tracheal obstruction.1–3 Most studies have reported ACC as tumour of the middle age (mean age 50 years), being extremely rare under the age of 30 years.1,2 However our patient presented at a relatively younger age of 25 years. Rigid bronchoscopy under GA is the conventional method for obtaining tissue samples for histology and assessment of the extent of stenosis and intraluminal spread of the tumour1,3 but it was not practicable in our case as the patient became severely breathless in supine position. There was also a potential risk for the development of fatal hypoxia due to airway obstruction as a result of bleeding, secretions, and oedema or from the bronchoscope itself.1 Our rationale behind opting for Chamberlains procedure was (a) to acquire biopsy without resorting to GA5 (b)
to access anterior mediastinum for tumour staging in order to establish surgical resectability5 and (c) to attain definite result (Chamberlain has a 100% specificity and 100% sensitivity with a morbidity and mortality rate of 16.1% and 1.6%, respectively).5 In conclusion, we were able to use Chamberlain mediastinotomy to biopsy the circumtracheal tumour under local anaesthesia. It proved to be a quick and efficient approach in our case and can be considered to biopsy tracheal lesions in patients who cannot tolerate general anaesthesia.
References 1. Maziak DE, Todd TR, Keshavjee SH, Winton TL, Van Nostrand P, Pearson FG. Adenoid cystic carcinoma of the airway: thirty-two-year experience. J Thorac Cardiovasc Surg 1996;112: 1522–31. 2. Webb BD, Walsh GL, Roberts DB, Sturgis EM. Primary tracheal malignant neoplasms: the University of Texas MD Anderson Cancer Center experience. J Am College Surg 2006;202: 237–46. 3. Meyers BF, Mathisen DJ. Management of tracheal neoplasms. Oncologist 1997;2:245–53. 4. Asai T, Shingu K. Airway management of a patient with tracheal stenosis for surgery in the prone position. Can J Anesth 2004;51:733–6. 5. Best LA, Munichor M, Ben-Shakhar M, Lemer J, Lichtig C, Peleg H. The contribution of anterior mediastinotomy in the diagnosis and evaluation of diseases of the mediastinum and lung. Ann Thorac Surg 1987;43:78–81.
515 CASE REPORTS
Heart, Lung and Circulation 2008;17:505–518
Chamberlain Mediastinotomy for Diagnosis of Adenoid Cystic Carcinoma of Trachea Saulat H. Fatimi, MD, FACS a , Rubina A. Sajwani, MBBS a , Nadeem Rizvi, MBBS b and Muhammad A. Javed, MD a Asmatullah Khan, MBBS a Usman Ahmad, MD a,∗ a
Department of Surgery, Division of Cardiothoracic Surgery, Aga Khan University, Karachi, Pakistan b Division of Chest Diseases, Jinnah Post Graduate Medical Center, Karachi, Pakistan
Primary tracheal tumours are extremely rare. Bronchoscopy is the standard diagnostic procedure of obtaining biopsy of a tracheal mass, however it becomes challenging if the obstructing lesion is placed distally along the trachea occluding greater than 90% of the airway. We report the case of a 25-year-old male who suffered from severe tracheal stenosis. The lesion was biopsied through a chamberlain mediastinotomy, under local and mask anaesthesia and was found to be primary adenoid cystic carcinoma. (Heart, Lung and Circulation 2008;17:505–518) © 2007 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved. Keywords. Chamberlain’s mediastinotomy; Anterior mediastinotomy; Adenoid cystic carcinoma; Trachea
Introduction
P
rimary tracheal malignancy has an incidence of <2 million persons per year.1 Malignant lesions rarely cause tracheal stenosis, but when present, adenoid cystic carcinoma (ACC) is the second most common histologic type.1,2 Rigid bronchoscopy under general anaesthesia (GA) is the conventional technique employed to biopsy,1–3 but management of airway in patients with severe tracheal obstruction is challenging for both the surgeon and the anaesthesiologist.4 We report our successful experience of establishing diagnosis of primary tracheal ACC in a patient with severe tracheal stenosis using chamberlain’s mediastinotomy under local and mask anaesthesia.
Case Presentation A 25-year-old nonsmoker male presented with progressively worsening dyspnoea for 2 years and exertional dyspnoea for 5 months. He was initially treated for asthma at another hospital but his condition soon deteriorated and he developed severe breathlessness on lying supine. Chest roentgenogram and thoracic computed tomographic scan showed a homogenous circumtracheal mass extending from lower part of trachea to the carina obstructing greater Received 11 February 2007; received in revised form 21 July 2007; accepted 30 July 2007; available online 10 October 2007 ∗ Corresponding author at: Department of Surgery, Yale University, Boyer Center for Molecular Medicine, Room 437, 295 Congress Ave, New Haven, CT 06510, USA. Tel.: +1 203 737 2289. E-mail address: [email protected] (U. Ahmad).
than 85% of the tracheal lumen. He was referred to our centre for an elective biopsy. The patient had no past history of tuberculosis or other medical comorbids including diabetes, hypertension, asthma, gastro-oesophageal reflux or allergy. Chest examination revealed grade IV wheezing and expiratory stridor. Chest wall retractions were also observed. Arterial blood gases in room air showed mild hypoxaemia and CO2 retention, with PaO2 = 75 mmHg, PaCO2 = 46 mmHg and oxygen saturation of 96%. Considering the patients’ inability to lie supine and to undergo intubation, Chamberlain’s mediastinotomy was carried out under local and inhalation anaesthesia, with the patient in a semi-recumbent position. After infiltrating the area with 2% xylocaine and epinephrine, an incision was made above the third rib in the second intercostal space on the right side. The superior vena cava, phrenic nerve, aorta and trachea were identified. The trachea was narrow and fibrotic. Multiple biopsy samples of tissue around the trachea and carina along with several paratracheal lymph nodes were taken. Frozen section was performed to ensure that the tissue obtained was a representative specimen. Histopathological examination showed the biopsy specimen to be positive for epithelial carcinoma. Final histopathological examination confirmed infiltrating ACC (Fig. 1). Post-operatively, the patient required 3 days to recover and had to be observed for a day as he was retaining CO2 . There were no major operative complications. Unfortunately further management was hindered as the patient failed to follow up.
© 2007 Australasian Society of Cardiac and Thoracic Surgeons and the Cardiac Society of Australia and New Zealand. Published by Elsevier Inc. All rights reserved.
1443-9506/04/$30.00 doi:10.1016/j.hlc.2007.07.008
516
Case Reports
CASE REPORTS
Heart, Lung and Circulation 2008;17:505–518
Fig. 1. Adenoid cystic carcinoma: H&E stain. (A) 20× and (B) 40×.
Discussion Neoplastic lesions are an extremely rare cause of tracheal stenosis.1 ACC is the second most common primary tracheal malignancy,1,2 and is characterised by slow growth and infiltrating nature often leading to late diagnosis. However it is frequently amenable to surgical resection with a good long term survival.1,2 The typical presenting symptoms are those of airway obstruction, i.e. wheezing, progressive dyspnoea, stridor, cough and haemoptysis so it is not uncommon for these symptoms to be mistaken for asthma.1,2 Chest X-ray in these patients is often normal,1,3 therefore CT scan (95% sensitive) is highly recommended to provide early detection of tracheal obstruction.1–3 Most studies have reported ACC as tumour of the middle age (mean age 50 years), being extremely rare under the age of 30 years.1,2 However our patient presented at a relatively younger age of 25 years. Rigid bronchoscopy under GA is the conventional method for obtaining tissue samples for histology and assessment of the extent of stenosis and intraluminal spread of the tumour1,3 but it was not practicable in our case as the patient became severely breathless in supine position. There was also a potential risk for the development of fatal hypoxia due to airway obstruction as a result of bleeding, secretions, and oedema or from the bronchoscope itself.1 Our rationale behind opting for Chamberlains procedure was (a) to acquire biopsy without resorting to GA5 (b)
to access anterior mediastinum for tumour staging in order to establish surgical resectability5 and (c) to attain definite result (Chamberlain has a 100% specificity and 100% sensitivity with a morbidity and mortality rate of 16.1% and 1.6%, respectively).5 In conclusion, we were able to use Chamberlain mediastinotomy to biopsy the circumtracheal tumour under local anaesthesia. It proved to be a quick and efficient approach in our case and can be considered to biopsy tracheal lesions in patients who cannot tolerate general anaesthesia.
References 1. Maziak DE, Todd TR, Keshavjee SH, Winton TL, Van Nostrand P, Pearson FG. Adenoid cystic carcinoma of the airway: thirty-two-year experience. J Thorac Cardiovasc Surg 1996;112: 1522–31. 2. Webb BD, Walsh GL, Roberts DB, Sturgis EM. Primary tracheal malignant neoplasms: the University of Texas MD Anderson Cancer Center experience. J Am College Surg 2006;202: 237–46. 3. Meyers BF, Mathisen DJ. Management of tracheal neoplasms. Oncologist 1997;2:245–53. 4. Asai T, Shingu K. Airway management of a patient with tracheal stenosis for surgery in the prone position. Can J Anesth 2004;51:733–6. 5. Best LA, Munichor M, Ben-Shakhar M, Lemer J, Lichtig C, Peleg H. The contribution of anterior mediastinotomy in the diagnosis and evaluation of diseases of the mediastinum and lung. Ann Thorac Surg 1987;43:78–81.