Ann Thorac Surg 2008;85:660-662. doi:10.1016/j.athoracsur.2007.07.088
© 2008 The Society of Thoracic Surgeons
Case Reports
Benign Esophageal Schwannoma Compressing the Trachea in Pregnancy
Shinjiro Mizuguchi, MD, PhDa,*,
Kiyotoshi Inoue, MD, PhDa,
Atsuo Imagawa, MDa,
Yoshinori Kitano, MD, PhDb,
Masao Kameyama, MD, PhDb,
Haruhiko Ueda, MD, PhDc,
Yasuhide Inoue, MD, PhDc
a Department of Thoracic Surgery, Bell Land General Hospital, Osaka, Japan
b Department of Surgery, Bell Land General Hospital, Osaka, Japan
c Department of Obstetrics and Gynecology, Bell Land General Hospital, Osaka, Japan
Accepted for publication July 27, 2007.
* Address correspondence to Dr Mizuguchi, Department of Thoracic Surgery, Bell Land General Hospital, 500-3 Higashiyama, Naka-ku, Sakai-city, Osaka, 599-8247, Japan (Email: m1293795{at}msic.med.osaka-cu.ac.jp).
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Abstract
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A rare case of esophageal schwannoma compressing the trachea in pregnancy is presented. A 29-year-old pregnant woman was hospitalized due to severe dyspnea. Imaging studies revealed a homogeneous tumor (8 cm in diameter) in the posterior mediastinum with compression of the lower trachea. After an uneventful cesarean section, the patient underwent a mini-axillary thoracotomy with video-assisted thoracic surgery. The tumor arose from within the muscular layers of the esophagus and was enucleated by gentle blunt dissection. Pathologic and immunohistochemical examinations revealed a benign esophageal schwannoma.
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Introduction
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Although neurogenic tumors are the most common primary tumors of the mediastinum, a benign schwannoma of the esophagus is rare. We present an extremely rare case of esophageal schwannoma compressing the trachea and the clinical and pathologic characteristics of patients with esophageal schwannoma reported in the literature.
A 29-year-old pregnant woman (gestation, 36 weeks) was hospitalized at our institution due to severe dyspnea after a mediastinal tumor was detected by imaging studies at another hospital. The dyspnea increased in the supine position. There was no hoarseness or dysphagia. No tumor markers were elevated. She was treated for bronchial asthma 1 year previously. A chest radiographic scan showed an abnormal shadow under the bifurcation of the trachea. A computed tomographic scan revealed a low-density mass (8 cm in diameter) posterior to the trachea. An elective cesarean section under spinal anesthesia was performed uneventfully on the second day after admission.
Enhanced computed tomography and magnetic resonance imaging revealed a homogeneous tumor in the posterior mediastinum with encasement around organs such as the trachea, esophagus, azygous vein, vertebrae, and aorta (Fig 1). Although bronchoscopic findings revealed severe stenosis of the carina with a smooth surface, bronchoscopy could pass the region of stenosis and intubation was considered possible. Esophagoscopy showed extrinsic compression from 19 to 27 cm from the incisors without mucosal ulceration. Ultrasonographic endoscopy visualized the mass lesion as hypoechoic and homogeneous, encasing the trachea. Although the origin of the tumor was not revealed by these examinations, esophageal leiomyoma was the most likely diagnosis without invasion of the trachea or aorta.
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Fig 1. Enhanced chest computed tomographic (A) coronal scan and (B) sagittal scan reveal a low-density mass surrounded by the trachea, esophagus, vertebrae, and aorta. The tumor compresses the trachea and thoracic esophagus.
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The patient underwent a mini-axillary thoracotomy with video-assisted thoracic surgery. The operation was started with ventilation of both lungs, because the left bronchus was displaced, and the double lumen tube was induced into the correct position with manipulation in the operative field. The tumor was found to arise from within the muscular layers of the esophagus, and extended from 6 cm on the cranial side of the azygous vein to 1 cm on the caudal side of the azygous vein. The esophageal muscular layer was divided and the tumor was enucleated by gentle blunt dissection (Fig 2). The esophagus was repaired using intermittent two-layer sutures due to a partial mucosal defect. A water-sealing test was performed. The distal esophagus was clamped and air was instilled into a proximally placed nasogastric tube. No bubbles escaped into the saline placed into the pleural cavity, indicating absence of any leak.
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Fig 2. Intraoperative appearance of the esophageal schwannoma arising from the thoracic esophagus. (SVC = superior vena cava.)
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The resected tumor was huge (8.0 x 7.5 x 6.0 cm) and had a yellowish cut-surface. Furthermore, the tumor was homogeneous and elastic hard with no hemorrhage or necrosis. Microscopically, the tumor was composed of compact bundles of spindle-shaped cells and mitosis was rare. Immunohistochemical staining was positive for S-100 and negative for CD34, desmin, c-kit and 
-smooth muscle actin. Histologic examination revealed a benign esophageal schwannoma. The patient recovered uneventfully and was discharged at 15 days after the surgery. She was currently well at 5 months after the treatment with no evidence of recurrence or tracheal stenosis.
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Comment
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The most common submucosal tumors of the digestive tract are leiomyomas, especially in the esophagus and stomach. Daimaru and colleagues [1] investigated 306 gastrointestinal spindle-cell tumor specimens by immunohistochemistry and identified 24 cases (7.8%) as schwannomas. Esophageal schwannoma is extremely rare, with only 30 reported cases. Reviews of previous reports [2–4] indicate that benign schwannomas are usually located in the upper esophagus and occur in middle-aged women. The most common symptom is dysphagia, and barium esophagograms show large, smooth, and polypoid-filling defects. Because there are no distinctive characteristics compared with other submucosal tumors, differentiation of schwannoma from other submucosal tumors is very difficult during preoperative examinations. Histologically, the diagnosis of schwannoma is confirmed immunohistochemically by the presence of S-100 protein and absence of histologic markers of smooth muscle, such as desmin [2–4]. In many cases, a benign esophageal schwannoma can be enucleated without the need for esophageal resection, and only one case with a huge esophageal schwannoma was reported to require esophagectomy [4].
There is only one previous case report of a benign schwannoma in the upper esophagus compressing the trachea [5]. Compression or invasion of the trachea sometimes occurs with malignant tumors, such as lung cancer, esophageal cancer, or thyroid cancer. However, compression of the trachea by benign mediastinal tumors is rare, because these tumors tend to extend into the pleural cavity. In the present case, the tumor was in the upper esophagus where it caused some airway compression or deviation. We considered that the tumor could not extend into the pleural cavity because it was surrounded by the azygous vein (right side), aorta (left side), and vertebrae (posterior side). Therefore, the tumor in our patient compressed the lower trachea, causing exertional dyspnea.
Some authors have reported the management of pregnant patients with a huge intrathoracic tumor (ie, Ewing type sarcoma, leiomyosarcoma) or anterior mediastinal tumor (ie, Hodgkin’s disease) by cesarean section [6–8]. In pregnancy, the physiologic changes of pregnancy may mask the symptoms, such as dyspnea. These diseases also make anesthetic management difficult due to the tracheal compression by the tumors. We present the first report of a benign esophageal schwannoma compressing the trachea in pregnancy. An elective cesarean section under spinal anesthesia was performed uneventfully, although we were prepared to use other methods, such as intubation, general anesthesia, or emergency thoracotomy.
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References
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- Daimaru Y, Kido H, Hashimoto H, Enjoji M. Benign schwannoma of the gastrointestinal tract: a clinicopathologic and immunohistochemical study Hum Pathol 1988;19:257-264.[Medline]
- Ohno M, Sugihara J, Miyamura K, et al. Benign schwannoma of the esophagus removed by enucleation: report of a case Surg Today 2000;30:59-62.[Medline]
- Saito R, Kitamura M, Suzuki H, Ogawa J, Sageshima M. Esophageal schwannoma Ann Thorac Surg 2000;69:1947-1949.[Abstract/Free Full Text]
- Park BJ, Carrasquillo J, Bains MS, Flores RM. Giant benign esophageal schwannoma requiring esophagectomy Ann Thorac Surg 2006;82:340-342.[Abstract/Free Full Text]
- Chen HC, Huang HJ, Wu CY, Lin TS, Fang HY. Esophageal schwannoma with tracheal compression Thorac Cardiovasc Surg 2006;54:555-558.[Medline]
- Szokol JW, Alspach D, Mehta MK, Parilla BV, Liptay MJ. Intermittent airway obstruction and superior vena cava syndrome in a patient with an undiagnosed mediastinal mass after cesarean delivery Anesth Analg 2003;97:883-884.[Abstract/Free Full Text]
- Crosby E. Clinical case discussion: anesthesia for Cesarean section in a parturient with a large intrathoracic tumour Can J Anaesth 2001;48:575-583.[Medline]
- Fernandez CL, Kuczkowski KM. Images in Anesthesia: intrathoracic leiomyosarcoma in a parturient presenting for Cesarean delivery Can J Anaesth 2003;50:1051.[Medline]
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Abstract
Introduction
