Successful Endoscopic Resection of Intramural Metastasis After Esophagectomy for Esophageal Cancer: A Case Report

• Introduction
• Case Report
• Discussion
• References

Carcinoma of the esophagus is often accompanied by intramural metastasis (IM) at the time of diagnosis, and the prognosis of patients with such metastasis is very poor. Here we report the case of a 60-year-old man who presented with a submucosal tumor in the wall of the cervical esophagus at 2 years after esophagectomy for thoracic esophageal cancer. The tumor was resected endoscopically and was histologically shown to be an IM of esophageal cancer. He has shown no recurrence and no metastasis during 2 years of follow-up after endoscopic resection and radiation therapy.

Introduction

Metastasis from a primary esophageal cancer to the adjacent esophageal or gastric wall is termed intramural metastasis (IM), and its presence has been reported to be an important factor for preoperative and postoperative evaluation of the prognosis [1] [2] [3] . The 5-year survival rate of patients with IM has been reported to be less than 10 % [2] [3] , and conventional radiotherapy or chemotherapy after surgery are ineffective in improving the prognosis [3]. It is difficult to determine an effective therapeutic modality for patients with IM, especially those with IM in the residual esophagus after esophagectomy. We report a rare case of IM in the residual esophagus that was successfully treated by endoscopic resection and radiation therapy.

Case Report

A 60-year-old man underwent radical esophagectomy along with 2-field dissection with extended superior mediastinal dissection for cancer of the thoracic esophagus, on December 6, 1994. A gastric tube was used for esophageal reconstruction, being inserted via the retrosternal route with the anastomosis in the neck. Macroscopically, two large elevated lesions were present in the middle third of the thoracic esophagus. The lesion on the anterior wall was cauliflower-like and 2.5 cm in size while the one on the posterior wall was polypoid and 3 cm in size with a slightly depressed unstained area on iodine staining (Figures [1 A, B]). Microscopic examination showed poorly differentiated squamous cell carcinoma with invasion to the proper muscle layer of the anterior wall and well differentiated squamous cell carcinoma with invasion to the adventitia of the posterior wall (Figures [1 C, D]). Although lymphatic and vascular permeation was not detected for either tumor, there was metastasis to the bilateral cardiac lymph nodes and paratracheal nodes. Postoperative radiation therapy was given to the supraclavicular and upper mediastinal area, with a total dose of 45 Gy. There was no recurrence and no metastasis to other organs during about 2 years of follow-up.

Endoscopic examination was performed in December 1996, and a submucosal tumor-like elevated lesion with a slight central depression was detected on the posterior wall about 19 cm from the incisors and about 3 to 4 cm above the esophagogastric anastomosis. Lugol dye technique showed normal staining over the tumor, except for a faintly stained central depression (Figures [2 A, B]). This tumor was thought to be in the submucosal layer because it had good mobility when gentle pressure was applied with a biopsy forceps.

Judging from the history of this patient and the appearance of the elevated lesion, the tumor was thought to be an IM of esophageal carcinoma. However, the biopsy specimen taken from the surface of the lesion showed normal squamous epithelium. Therefore, the patient was admitted to our hospital for endoscopic resection to make a definite diagnosis and for treatment of the tumor. There was no lymphadenopathy seen in the cervical or supraclavicular regions on ultrasonography and computed tomography.

Endoscopic resection using a Pneumo-activate endoscopic variceal ligation (EVL) device (Sumitomo Bakelite Co. Ltd., Tokyo, Japan) was performed in January 1997. The snare was closed outside the transparent plastic cap. The lesion was sucked into the cap and snared tightly by injection of air into the device. Then the submucosal tumor was resected by electrocautery and the specimen was easily retrieved by holding it firmly inside the cap. Neither bleeding nor perforation occurred and the postoperative course was uneventful (Figure [2 C]). The resected specimen measured 1.5 cm ×1.5 cm in size and included part of the proper muscle layer. Histological examination revealed IM of well differentiated squamous cell carcinoma in the submucosal layer (Figures [3 A, B]). Radiation therapy was given to the supraclavicular region with a total dose of 20 Gy, and intraesophageal radiotherapy was performed three times using a remote afterloading system with a dose of 5 Gy. There has been no recurrence and no metastasis to other organs during 2 years of follow-up (Figure [2 D]).

Discussion

Carcinoma of the esophagus is often accompanied by IM at the time of diagnosis. In previous studies the incidence of IM has ranged from 9.0 % to 16.7 % in resected esophagi [1] [2] [3] [4] . IM occurs in the esophagus distally or proximally to the primary carcinoma and even affects the stomach in 1.7 % to 5.5 % of cases [1] [2] [4] . Several studies on the distance between IM and the primary tumor have shown that it sometimes exceeds 10 cm [2] [3] . Thus, care must be taken to make an early diagnosis, by periodic endoscopic examination, of both anastomotic recurrence and IM in the residual esophagus after surgery.

Takubo et al. [2] reported that IM showed erosions or ulceration histologically in 42.9 % out of 42 lesions and stated that IM with erosions or ulceration was easily differentiated from submucosal tumors, which rarely had such changes. This may be an important finding to use in barium studies and endoscopic examination for separating IM from other submucosal tumors. Our patient's IM lesion was the small dome-like mass covered with epithelium that showed normal iodine staining and had a superficial erosion, which suggested that it was indeed IM. In recent years endoscopic ultrasonography (EUS) has come to be regarded as the most useful tool available for the diagnosis of submucosal tumors [5]. However, tumors that are difficult to characterize or show an increase in size still require removal for histological diagnosis. Endoscopic submucosal tumorectomy has been reported to be useful as both a diagnostic and a therapeutic procedure. For treating selected submucosal tumors of the esophagus, several procedures have previously been reported, including snare polypectomy [6], pur ethanol topical injection [7], endoscopic incisional enucleation [8], and aspiration lumpectomy [9]. Kajiyama et al. [9] modified aspiration mucosectomy using the cap-fitted panendoscope, which was originally developed by Inoue and Endo [10], and applied it to the resection of esophageal leiomyomas. We used a Pneumo-activate EVL device for aspiration tumorectomy. This method does not require a Teflon tube, and the snare band could be closed easily by injection of air into the device.

In previous studies the majority of esophageal submucosal tumors resected endoscopically were benign lesions, such as leiomyoma, granular cell tumor, neurogenic tumor, lipoma, and cyst [6] [7] [8] [9] . To our knowledge, the present case is the first description of successful endoscopic treatment of IM arising from esophageal cancer. Although cervical esophagectomy or laryngectomy with radical neck dissection may be required for this case, these procedures have the disadvantages of severe surgical stress and loss of laryngeal function. In addition, the prognosis of patients with esophageal cancer accompanied by IM is much poorer than that of those without IM because of the high incidence of lymph node metastasis and lymphatic invasion [1] [2] [3] [4] . For these reasons we selected endoscopic treatment and additional radiation therapy as an alternative to surgery. The present case serves to demonstrate that careful and periodic endoscopic follow-up is needed after resection of esophageal cancer to allow early detection of IM as well as second cancers of the head and neck. Aggressive and carefully targeted treatment of patients with recurrent disease may offer them a chance of long-term survival.

References

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S. Tamura, M.D.

Dept. of Surgery, Kinki Central Hospital of the Mutual Aid Association of Public School Teachers

3-1 Kurumazuka, Itami, Hyogo 664-0872, Japan

Fax: Fax:+ 81-727-79-1567

References

• 1 Saito T, Iizuka T, Kato H, Watanabe H. Esophageal carcinoma metastatic to the stomach. A clinicopathologic study of 35 cases.  Cancer. 1985;  56 2235-2241
  PubMedSearch in Google Scholar
• 2 Takubo K, Sasajima K, Yamashita K, et al. Prognostic significance of intramural metastasis in patients with esophageal carcinoma.  Cancer. 1990;  65 1816-1819
  PubMedSearch in Google Scholar
• 3 Nishimaki T, Suzuki T, Tanaka Y, et al. Intramural metastasis from thoracic esophageal cancer: local indicator of advanced disease.  World J Surg. 1996;  20 32-37
  CrossrefPubMedSearch in Google Scholar
• 4 Maeta M, Kondo A, Shibata S, et al. Esophageal cancer associated with multiple cancerous lesions: clinicopathologic comparisons between multiple primary and intramural metastatic lesions.  Gastroenterol Jpn. 1993;  28 187-192
  PubMedSearch in Google Scholar
• 5 Murata Y, Yoshida M, Akimoto S, et al. Evaluation of endoscopic ultrasonography for the diagnosis of submucosal tumors of the esophagus.  Surg Endosc. 1988;  2 51-58
  CrossrefPubMedSearch in Google Scholar
• 6 Benedetti G, Sablich R, Bonea M, Mariuz S. Fiberoptic endoscopic resection of symptomatic leiomyoma of upper esophagus.  Acta Chir Scand. 1990;  156 807-808
  PubMedSearch in Google Scholar
• 7 Eda Y, Asaki S, Yamagata L, et al. Endoscopic treatment for submucosal tumors of the esophagus: studies in 25 patients.  Gastroenterol Jpn. 1990;  25 411-416
  PubMedSearch in Google Scholar
• 8 Hyun J H, Jeen Y T, Chun H J, et al. Endoscopic resection of submucosal tumor of the esophagus: results in 62 patients.  Endoscopy. 1997;  29 165-170
  Thieme ConnectPubMedSearch in Google Scholar
• 9 Kajiyama T, Sakai M, Torii A, et al. Endoscopic aspiration lumpectomy of esophageal leiomyomas derived from the muscularis mucosae.  Am J Gastroenterol. 1995;  90 417-422
  PubMedSearch in Google Scholar
• 10 Inoue H, Endo M. A new simplified technique of endoscopic esophageal mucosal resection using a cap-fitted panendoscope.  Surg Endosc. 1993;  6 264-265
  PubMedSearch in Google Scholar

S. Tamura, M.D.

Dept. of Surgery, Kinki Central Hospital of the Mutual Aid Association of Public School Teachers

3-1 Kurumazuka, Itami, Hyogo 664-0872, Japan

Fax: Fax:+ 81-727-79-1567