Ann Thorac Surg 2006;81:2283-2285
© 2006 The Society of Thoracic Surgeons
Case report
Huge Benign Mesenchymoma in Pharynx-Esophagus
Jun Li, MD
*
,
Alfred Omo, MD,
Ligang Liu, MD,
Lisi Liu, MD,
Yinxiong Tang, MD,
Tiecheng Pan, MD
Department of Cardiothoracic Surgery, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China
Accepted for publication August 22, 2005.
* Address correspondence to Dr Li, Tongji Hospital, Department of Cardiothoracic Surgery, Wuhan, 430030 China (Email: junliulmde{at}yahoo.com.cn).
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Abstract
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Benign mesenchymoma is an uncommon neoplastic disease and its occurrence in pharynx-esophagus is even more rarely reported. A successful case operation is reported. The origin of this tumor was in the pharynx-esophagus, and complete excision was achieved through a laterocervical approach.
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Introduction
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Mesenchymoma was first introduced by Klein [1] in 1932 to describe a primitive mesenchymal neoplasm that microscopically resembled undifferentiated mesenchyme. In 1962, Le Ber and Stout [2] described 39 cases of benign mesenchymoma occurring in various anatomic locations. The majority of them presented in the upper and lower extremities, kidneys, trunk, and perirenal areas. During the last 13 years, we have observed and surgically treated three cases of huge benign mesenchymoma in pharynx-esophagus, and we have previously reported two cases in the Chinese literature [3, 4]. Here we report our new case because there are few such cases reported in the English medical literature.
A 36-year-old man was admitted into our department in January 2005 with a chief complaint of pain in the pharynx and chest after swallowing for 4 years, which was associated with dysphagia for 1 year.
Physical examinations showed no abnormalities except a palpable hard mass, approximately 4 x 4 x 6 cm in size on the left lateral side of the neck. Laboratory results were within normal range. Barium swallow examination suggested a possible malignant change in the upper portion of the esophagus. An upper gastrointestinal endoscopy revealed an irregular protruded mass occupying the esophageal lumen 18 to 26 cm from the canine. With the experience from the previous two cases [3, 4], the diagnosis of esophageal benign mesenchymoma was established. Under general anesthesia a left laterocervical incision and longitudinal esophagotomy was performed. A yellowish-white lobular mass held by a pedicle was observed, with its base located on the left lateral wall of the esophagus. The complete excision was performed, and the size of the removed tumor specimen measured 10 x 4.5 x 6 cm (Fig 1), with smooth surfaces but a hard texture.
Pathologic examination revealed a benign mesenchymoma of the upper portion of the esophagus (Fig 2). The patient recovered well after surgery and was discharged 12 days after the operation. The patient was free of any complaints 6 months after the surgery.
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Fig 2. Benign mesenchymoma composed of fibrous, mature fat, and muscle tissue. (Hematoxylin and eosin; x230.)
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Comment
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In 1932, Klein [1] first introduced the term mesenchymoma. In 1948, Stout [5] further defined this uncommon tumor as any mass composed of at least two unrelated mesenchymal elements other than fibrous tissues. The tumor is divided into benign and malignant types based on its level of differentiation. The most common mesenchymal elements include fat, blood vessels, lymphoid, smooth muscles, skeletal muscles, serous tissues, and islands of cartilage and bone tissues.
The benign mesenchymoma is most commonly seen in the extremities of the limbs, in the retroperitoneum, and it is rarely seen in the neck region, and even more rarely seen in the pharynx-esophagus. In 1978 Bures and Barnes [6] reported 18 cases of benign mesenchymoma presented in the head and neck region, and most of them were located on the subcutaneous layer of the skin, the tongue base of mouth cavity, the epiglottis, the trachea, and the esophagus. The majority of them were huge subcutaneous or submembranous granular masses. In our three cases, the tumors had solitary pedicles and originated from the lateral wall of the pharynx with their bodies hung down inside the esophageal lumen by their gravity. Because the tumor tissues had peculiar fast bulging growth characteristics, it blocked esophageal lumen down toward the cardia. The tumors were polyp-like and all had developed from the submucosal layer of the lateral wall of pharynx. The biggest tumor of the three cases measured 40 x 8 x 5 cm. The cut open surface of all three tumors revealed a yellowish-white solid tissue. Pathologic characteristics included fibrous tissues, large amounts of fat tissues, blood vessels, and serous components, which were similar to those reported by Enzinger and Zhang [7] in 1988.
There is no difference in either gender or age of the patients who have benign mesenchymoma. Our three cases were all male and older than 30 years of age. They had no significant symptoms in their early stages, but later developed dysphagia as the tumor became larger. The complication of melena was common because there was bleeding from the necrotic surfaces of the tumors. Two of our previously reported cases [3, 4] also presented as general weight lost, anemia, and malnutrition. Esophageal barium swallow, computed tomographic scans, and magnetic resonance scans revealed the size and location of the tumor inside the esophagus. These gave us a clue to suspect possible neoplastic changes occurring in the pharynx-esophagus.
Benign mesenchymoma in the pharynx-esophagus usually have their solitary pedicles attached to the lateral wall of pharynx. With pharyngoscopy and upper gastrointestinal endoscopy we can clearly see the precise locations of the base of these pedicles. Therefore the diagnosis of this rare disease requires combined analysis of its clinical presentations, endoscopies, and pathologic examinations. Meanwhile the benign mesenchymoma needs to be carefully differentiated from leiomyoma, lipoma, and malignant mesenchymoma. In the last case, pathologic nuclear fission could be seen under the microscope, the tumor easily reoccurred, and there was early metastasis with a poor prognosis.
There are limited experience and reports on surgical treatment of huge benign mesenchymoma of pharynx-esophagus. In our first case [3], due to the limited experience of this rare disease, diagnosis of leiomyoma of esophagus was given at the beginning. During the operation we could not remove the tumor through routine left thoracotomy. Since the pedicle originated from the upper portion, and laterocervical incision was needed for complete excision. With the accumulation of experience from the latter two cases, now we have more knowledge about this rare disease. When the diagnosis of benign mesenchymoma in pharynx-esophagus is confirmed, we believe that a laterocervical approach is an ideal way for excision of the tumor. After longitudinal esophagotomy, special attention must be paid to pull the body of the tumor out of esophagus before resection of its pedicle in order to avoid tearing the tumor from the pedicle and sliding down the lower portion of esophagus.
Usually patients with benign mesenchymoma have very smooth recovery course after operation, however Bures reported [6] that the recurrence rate was about 20% in patients with benign mesenchymoma located in head and neck regions. This was because that the tumor has no clear margin with its surrounding tissues, it was partially encapsulated and incomplete excision was thus performed due to this reason. In our follow-up data, there are no recurrences in all the three cases and we think that complete excision of the bases of tumor pedicles is the key to avoid this complication.
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References
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- Klein W. Mesenchymoma J Med Soc NJ 1932;29:774-778.
- Le Ber MS, Stout AP. Benign mesenchymomas in children Cancer 1962;15:598-605.[Medline]
- Liu LS, Zhen HT. Submucous membrane huge benign mesenchymoma in pharynx-esophagus Acta Univ Med Tongji 1999;28:561-562.
- Tao YL, Liu LS, Cui YH, Tang YX. Huge benign mesenchymoma in the pharynx-esophagus Lin Chuang Er Bi Yan Hou Ke Za Zhi 2000;14:549-550.[Medline]
- Stout AP. Mesenchymoma, the mixed tumor of mesenchymal derivatives Ann Surg 1948;127:278-290.[Medline]
- Bures C, Barnes L. Benign mesenchymomas of the head and neck Arch Pathol Lab Med 1978;102:237-241.[Medline]
- Enzinger FM, Zhang RY. Plexiform fibrohistiocytic tumor presenting in children and young adults. An analysis of 65 cases Am J Surg Pathol 1988;12:818-826.[Medline]
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Abstract
Introduction
