Gen Thorac Cardiovasc Surg DOI 10.1007/s11748-014-0469-8
CASE REPORT
Adenoid cystic carcinoma of the lower trachea treated by resection of 11 of 18 rings of the total length: report of a case Hiroaki Nomori • Masaru Abe • Hiroshi Sugimura Akihiko Takeshi
•
Received: 22 July 2014 / Accepted: 28 August 2014 Ó The Japanese Association for Thoracic Surgery 2014
Abstract We report the case of a 63-year-old woman with adenoid cystic carcinoma of the lower trachea treated by resection of 11 of the 18 cartilaginous rings (61 %) of the total length. The little remaining membranous portion of the carina was sewn up to create a margin for anastomosis. The anastomotic sites could be approximated by the mobilization of the cervical trachea and the left main bronchus, pulling across the traction sutures, and anteflexion of the neck. The patient’s postoperative course was uneventful without any complications associated with anastomosis. Because both the proximal and distal margins showed microscopic tumors, radiation therapy was performed with 50 Gy 2 months after surgery. The patient has a good social life without recurrence 20 months after surgery. Keywords Adenoid cystic carcinoma Lower trachea Tracheoplasty
Introduction Adenoid cystic carcinoma (ACC) of the trachea is well known to extend along the long axis, necessitating difficult anastomosis especially for tumors in the lower trachea because of high tension at the anastomosis [1], but it can be cured with radical surgery and postoperative irradiation [2, 3]. Here, we present a case of ACC of the lower trachea that required the resection of 11 of 18 cartilaginous rings (61 %) of the total length. H. Nomori (&) M. Abe H. Sugimura A. Takeshi Department of Thoracic Surgery, Kameda Medical Center, 929 Higashi-cho, Kamogawa, Chiba 296-8602, Japan e-mail: [email protected]
Case report The patient was a 63-year-old woman who complained worsening dyspnea and hemosputum. Computed tomography (CT) showed a stricture of the lower trachea by the sloping tumor (Fig. 1). While preoperative bronchoscopy was not available due to severe airway stenosis, a coronal CT scan showed that the trachea had 18 cartilaginous rings in total length and the tumor seemed to be located at the 11th ring of trachea to the right main bronchus. Because the tumor was expected to be ACC from the findings of CT, the treatment was scheduled to resect the tumor grossly by a sleeve tracheal resection to release the airway stenosis followed by postoperative radiation therapy. We consider that a combined carinal resection should be avoided because of its high risk of anastomotic failure in a case with such a long length of tracheal resection. In December 2012, an awake intubation was conducted because of severe airway stenosis, followed by general anesthesia. Posterolateral thoracotomy was performed via the fourth intercostal space. The operative findings showed the tumor to be located longer than the CT findings toward the proximal site, which extended over two-thirds of the upper mediastinum. The trachea was divided at the distal site of the tumor, followed by intubation into the left main bronchus from the operative field. Because the trachea was modestly flexible, we assessed that a gross resection of tumor by trachea sleeve resection would be available. The trachea was then divided at the proximal site of the tumor. Because the surgical margin at the distal site showed a grossly remained tumor, 2 rings of the distal site was additionally resected, resulting in resection of a major portion of the mediastinal trachea (Fig. 2). Intraoperative frozen section was not examined because a further resection was impossible. Because there was little remaining
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Gen Thorac Cardiovasc Surg
Fig. 1 Coronal CT view showing stenosis of lower trachea by tumor, which is indicated by an arrow
Fig. 3 Top sewing up the membranous portion of carina. Bottom interrupted sutures were placed at whole circumference. Traction sutures were also placed
Fig. 4 Bronchoscopy finding 4 months after surgery Fig. 2 Operative finding showing that a major portion of the mediastinal trachea is resected. SVC superior vena cava
margin of the membranous portion at the carina, approximately 1 cm was sewn up using 2 stitches to construct an anastomotic margin (Fig. 3). The mobilization procedures were as follows: (1) the cervical trachea was manually mobilized up to the cricoid; (2) the left main bronchus was mobilized down to the second carina; and (3) the mediastinal pleura around the right upper hilum was divided.
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Interrupted anastomotic sutures using 4-0 polydioxanone were placed around the entire circumference. The anastomotic sutures were ligated all together, while the neck was anteflexed and the traction sutures placed at both sites were pulled across each other to approximate the anastomosis. The intercostal muscle flap was wrapped at the lateral side of anastomosis. After surgery, the patient was maintained in a position with anteflexion of the neck for a week. The postoperative course was uneventful. Bronchoscopy 4 months after
Gen Thorac Cardiovasc Surg
Fig. 5 Macroscopic findings of the resected materials. Left coronal section of the first resected trachea, showing 9 rings. Right axial section of the additionally resected trachea with 2 rings
surgery showed neither stenosis nor granulation, regardless of slight flattening (Fig. 4). The resected material contained 11 cartilaginous rings in total, which comprised 61 % of the 18 rings of the total length (Fig. 5). The pathological diagnosis was ACC with metastases in two lymph nodes near the tumor. Because both the proximal and distal margins showed microscopic tumors, radiation therapy was performed with 50 Gy 2 months after surgery. The patient now has a good social life without recurrence 20 months after surgery.
Discussion We chose the present treatment as an emergency life-saving from severe stenosis of the central airway. To do it, the resection of stenosis area was the highest importance regardless of microscopically positive margin. Because it has been reported that the ACC of trachea is sensitive to radiation therapy [4, 5], we expected that postoperative radiation would cure the microscopically residual tumor. Two points are of note regarding the technique in the present case. The first is the resected length and area, i.e., 61 % of the lower trachea. It has been reported that a half or more of the trachea can be resected and reconstructed. In autopsy studies, Grillo et al. [6] demonstrated that 58 % of the trachea could be successfully resected and approximated. However, the most significant difference between their experiments and the present case was the following: they divided the trachea 2 cm below the cricoid and then excised the distal trachea, which was mainly the upper trachea, while in the present case, the lower trachea was
resected. The approximation of anastomotic sites is more difficult for the lower trachea than for the upper trachea because of the following: the distal anastomotic site in the plasty for upper tracheal resection is mediastinal trachea, which is easy to mobilize, while that for lower tracheal resection is close to lung hilum, which is hard to mobilize. The second point is that plasty of the membranous portion of the carina was performed to create sufficient anastomosis margin in the present case. If this was not done, the carina itself should have been sutured, which might have caused granulation stenosis at the carina by damage to the cartilage by a suture—which would have resulted in serious airway stenosis. To avoid this, we plicated the membranous portion of the carina to create a cuff, which made the anastomosis success. While the postoperative course was uneventful, we reflect the present procedure on the following: the tension at the anastomosis could be reduced by saving the resection of trachea in each one ring at both proximal and distal sites, of which the extent of residual tumor would not be significantly different with that after the present procedure. However, to the best of our knowledge, resection of 61 % of the total length of the lower trachea is the largest ever reported and might be close to the limit of tracheoplasty for the lower trachea. Using the procedures described herein, we believe that this could be feasible. Conflict of interest The authors have no conflicts of interest or financial ties to disclose.
References 1. Nomori H, Kaseda S, Kobayashi K, et al. Adenoid cystic carcinoma of the trachea and main-stem bronchus. J Thorac Cardiovasc Surg. 1988;96:271–7. 2. Pearson FG, Todd TRJ, Cooper JD. Experience with primary neoplasms of the trachea and carina. J Thorac Cardiovasc Surg. 1984;88:511–8. 3. Perelman MI, Kovoleva N, Birjukov J, et al. Primary tracheal tumors. Sem Thorac Cardiovasc Surg. 1996;8:400–2. 4. Maziak DE, Todd TR, Keshavjee SH, Winton TL, Van Nostrand P, Pearson FG. Adenoid cystic carcinoma of the airway: thirtytwo-year experience. J Thorac Cardiovasc Surg. 1996;112:1522–31. 5. Bonner Millar LP, Stripp D, Cooper JD, Both S, James P, Rengan R. Definitive radiotherapy for unresected adenoid cystic carcinoma of the trachea. Chest. 2012;141:1323–6. 6. Grillo HC, Dignan EF, Miura T. Extensive resection and reconstruction of mediastinal trachea without prosthesis or graft. An anatomical study in man. J Thorac Cardiovasc Surg. 1964;48:741–9.
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CASE REPORT
Adenoid cystic carcinoma of the lower trachea treated by resection of 11 of 18 rings of the total length: report of a case Hiroaki Nomori • Masaru Abe • Hiroshi Sugimura Akihiko Takeshi
•
Received: 22 July 2014 / Accepted: 28 August 2014 Ó The Japanese Association for Thoracic Surgery 2014
Abstract We report the case of a 63-year-old woman with adenoid cystic carcinoma of the lower trachea treated by resection of 11 of the 18 cartilaginous rings (61 %) of the total length. The little remaining membranous portion of the carina was sewn up to create a margin for anastomosis. The anastomotic sites could be approximated by the mobilization of the cervical trachea and the left main bronchus, pulling across the traction sutures, and anteflexion of the neck. The patient’s postoperative course was uneventful without any complications associated with anastomosis. Because both the proximal and distal margins showed microscopic tumors, radiation therapy was performed with 50 Gy 2 months after surgery. The patient has a good social life without recurrence 20 months after surgery. Keywords Adenoid cystic carcinoma Lower trachea Tracheoplasty
Introduction Adenoid cystic carcinoma (ACC) of the trachea is well known to extend along the long axis, necessitating difficult anastomosis especially for tumors in the lower trachea because of high tension at the anastomosis [1], but it can be cured with radical surgery and postoperative irradiation [2, 3]. Here, we present a case of ACC of the lower trachea that required the resection of 11 of 18 cartilaginous rings (61 %) of the total length. H. Nomori (&) M. Abe H. Sugimura A. Takeshi Department of Thoracic Surgery, Kameda Medical Center, 929 Higashi-cho, Kamogawa, Chiba 296-8602, Japan e-mail: [email protected]
Case report The patient was a 63-year-old woman who complained worsening dyspnea and hemosputum. Computed tomography (CT) showed a stricture of the lower trachea by the sloping tumor (Fig. 1). While preoperative bronchoscopy was not available due to severe airway stenosis, a coronal CT scan showed that the trachea had 18 cartilaginous rings in total length and the tumor seemed to be located at the 11th ring of trachea to the right main bronchus. Because the tumor was expected to be ACC from the findings of CT, the treatment was scheduled to resect the tumor grossly by a sleeve tracheal resection to release the airway stenosis followed by postoperative radiation therapy. We consider that a combined carinal resection should be avoided because of its high risk of anastomotic failure in a case with such a long length of tracheal resection. In December 2012, an awake intubation was conducted because of severe airway stenosis, followed by general anesthesia. Posterolateral thoracotomy was performed via the fourth intercostal space. The operative findings showed the tumor to be located longer than the CT findings toward the proximal site, which extended over two-thirds of the upper mediastinum. The trachea was divided at the distal site of the tumor, followed by intubation into the left main bronchus from the operative field. Because the trachea was modestly flexible, we assessed that a gross resection of tumor by trachea sleeve resection would be available. The trachea was then divided at the proximal site of the tumor. Because the surgical margin at the distal site showed a grossly remained tumor, 2 rings of the distal site was additionally resected, resulting in resection of a major portion of the mediastinal trachea (Fig. 2). Intraoperative frozen section was not examined because a further resection was impossible. Because there was little remaining
123
Gen Thorac Cardiovasc Surg
Fig. 1 Coronal CT view showing stenosis of lower trachea by tumor, which is indicated by an arrow
Fig. 3 Top sewing up the membranous portion of carina. Bottom interrupted sutures were placed at whole circumference. Traction sutures were also placed
Fig. 4 Bronchoscopy finding 4 months after surgery Fig. 2 Operative finding showing that a major portion of the mediastinal trachea is resected. SVC superior vena cava
margin of the membranous portion at the carina, approximately 1 cm was sewn up using 2 stitches to construct an anastomotic margin (Fig. 3). The mobilization procedures were as follows: (1) the cervical trachea was manually mobilized up to the cricoid; (2) the left main bronchus was mobilized down to the second carina; and (3) the mediastinal pleura around the right upper hilum was divided.
123
Interrupted anastomotic sutures using 4-0 polydioxanone were placed around the entire circumference. The anastomotic sutures were ligated all together, while the neck was anteflexed and the traction sutures placed at both sites were pulled across each other to approximate the anastomosis. The intercostal muscle flap was wrapped at the lateral side of anastomosis. After surgery, the patient was maintained in a position with anteflexion of the neck for a week. The postoperative course was uneventful. Bronchoscopy 4 months after
Gen Thorac Cardiovasc Surg
Fig. 5 Macroscopic findings of the resected materials. Left coronal section of the first resected trachea, showing 9 rings. Right axial section of the additionally resected trachea with 2 rings
surgery showed neither stenosis nor granulation, regardless of slight flattening (Fig. 4). The resected material contained 11 cartilaginous rings in total, which comprised 61 % of the 18 rings of the total length (Fig. 5). The pathological diagnosis was ACC with metastases in two lymph nodes near the tumor. Because both the proximal and distal margins showed microscopic tumors, radiation therapy was performed with 50 Gy 2 months after surgery. The patient now has a good social life without recurrence 20 months after surgery.
Discussion We chose the present treatment as an emergency life-saving from severe stenosis of the central airway. To do it, the resection of stenosis area was the highest importance regardless of microscopically positive margin. Because it has been reported that the ACC of trachea is sensitive to radiation therapy [4, 5], we expected that postoperative radiation would cure the microscopically residual tumor. Two points are of note regarding the technique in the present case. The first is the resected length and area, i.e., 61 % of the lower trachea. It has been reported that a half or more of the trachea can be resected and reconstructed. In autopsy studies, Grillo et al. [6] demonstrated that 58 % of the trachea could be successfully resected and approximated. However, the most significant difference between their experiments and the present case was the following: they divided the trachea 2 cm below the cricoid and then excised the distal trachea, which was mainly the upper trachea, while in the present case, the lower trachea was
resected. The approximation of anastomotic sites is more difficult for the lower trachea than for the upper trachea because of the following: the distal anastomotic site in the plasty for upper tracheal resection is mediastinal trachea, which is easy to mobilize, while that for lower tracheal resection is close to lung hilum, which is hard to mobilize. The second point is that plasty of the membranous portion of the carina was performed to create sufficient anastomosis margin in the present case. If this was not done, the carina itself should have been sutured, which might have caused granulation stenosis at the carina by damage to the cartilage by a suture—which would have resulted in serious airway stenosis. To avoid this, we plicated the membranous portion of the carina to create a cuff, which made the anastomosis success. While the postoperative course was uneventful, we reflect the present procedure on the following: the tension at the anastomosis could be reduced by saving the resection of trachea in each one ring at both proximal and distal sites, of which the extent of residual tumor would not be significantly different with that after the present procedure. However, to the best of our knowledge, resection of 61 % of the total length of the lower trachea is the largest ever reported and might be close to the limit of tracheoplasty for the lower trachea. Using the procedures described herein, we believe that this could be feasible. Conflict of interest The authors have no conflicts of interest or financial ties to disclose.
References 1. Nomori H, Kaseda S, Kobayashi K, et al. Adenoid cystic carcinoma of the trachea and main-stem bronchus. J Thorac Cardiovasc Surg. 1988;96:271–7. 2. Pearson FG, Todd TRJ, Cooper JD. Experience with primary neoplasms of the trachea and carina. J Thorac Cardiovasc Surg. 1984;88:511–8. 3. Perelman MI, Kovoleva N, Birjukov J, et al. Primary tracheal tumors. Sem Thorac Cardiovasc Surg. 1996;8:400–2. 4. Maziak DE, Todd TR, Keshavjee SH, Winton TL, Van Nostrand P, Pearson FG. Adenoid cystic carcinoma of the airway: thirtytwo-year experience. J Thorac Cardiovasc Surg. 1996;112:1522–31. 5. Bonner Millar LP, Stripp D, Cooper JD, Both S, James P, Rengan R. Definitive radiotherapy for unresected adenoid cystic carcinoma of the trachea. Chest. 2012;141:1323–6. 6. Grillo HC, Dignan EF, Miura T. Extensive resection and reconstruction of mediastinal trachea without prosthesis or graft. An anatomical study in man. J Thorac Cardiovasc Surg. 1964;48:741–9.
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