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Long-term survival after thoracoscopic enucleation of a gastrointestinal stromal tumor arising from the esophagus

Long-term survival after thoracoscopic enucleation of a gastrointestinal stromal tumor arising... The goal of surgical treatment for gastrointestinal stromal tumor (GIST) is the complete resection of the tumor. A 62-year-old male had a clearly distinguishable mass having a smooth surface at the right side of the lower esophagus by computed tomography. Thoracoscopic resection of the tumor was performed. Immunohistochemical analysis showed that the tumor was positive for c-KIT and CD34 without mitosis, and diagnosed to be a low-risk GIST. At 6 years after surgery, the patient survived without recurrence. This study described the long-term surviving patient without the recurrence of tumor after the thoracoscopic resection of an esophageal GIST. INTRODUCTION Gastrointestinal stromal tumors (GISTs) in the esophagus are extremely rare. Reliable surgical therapy for GIST is the complete tumor resection. In principle, the resection of involved organ has been recommended for preserving physiological functions, and lymph-node dissection is unnecessary. This study described a patient who underwent the thoracoscopic enucleation of an esophageal GIST and survived without the recurrence of tumor for 6 years after surgery. CASE REPORT A 62-year-old male was found to have a well-demarcated mass with a smooth surface at the right side of the lower esophagus by chest computed tomography (CT) upon medical check-up (Fig. 1). Routine laboratory data and the serum levels of tumor markers were within normal limits. Positron emission tomography (PET) revealed the Figure 1: Chest CT of a 62-year-old male. The white arrow head indicates a clearly distinguishable mass having a smooth surface at the right side of the loweresophagus. accumulation of F-fluoro-2-deoxyglucose (FDG) at the tumor. Received: September 30, 2014. Revised: December 22, 2014. Accepted: December 29, 2014 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2015. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com 1 2 | Thoracoscopic resection of an esophageal GIST Figure 2: Endoscopic examination and endoscopic ultrasonography. (a) Tumor with a normally appearing mucosa was located 40 cm from the incisor teeth. (b) Hypoechoic submucosal tumor (asterisk) with annular localization, arising from the submucosal layer. The maximum standardized uptake value was 4.0, increasing to 4.8 in the late phase. Upper gastrointestinal endoscopy revealed no dis- tinct mucosal abnormalities or no evidence of tumors. Endoscopic ultrasonography showed a mass ∼2 cm in diameter arising in the third layer 40 cm from the incisor teeth (Fig. 2a and b). These find- ings diagnosed the lesion as an esophageal submucosal tumor with suspecting an esophageal leiomyoma or GIST. Being under general endotracheal double-lumen anesthesia with split-lung ventilation, the patient was allowed to lie on the healthy side (left) and posture lateral decubitus. A 6-cm long access incision was made at the pos- terior axillary line in the 10th intercostal space. Two 10.5-mm ports were made at the seventh intercostal space in the anterior axillary line and other at the same intercostal space in the posterior axillary line. After no adhesion in the thoracic cavity was observed, the pul- monary ligament was dissected and the lower lobe of the right lung was moved anteriorly. A tumefactive lesion was found in the lower esophagus. An incision was made in the mediastinal pleura, and Figure 3: Thoracoscopic view of the tumor. The image shows bulging through the the mass was confirmed at the outer longitudinal muscle of the right thoracic cavity. The black dash line and asterisk indicate the GIST; D, the lower esophagus. The tumor was easily removed from the sur- diaphragm; VD, the vertebral body. rounding tissue by a tumor enucleation procedure. No esophageal mucosa was injured intraoperatively. The operation time was 118 min, and the blood loss was 11 g (Fig. 3). Macroscopic examin- ation found that the removed white parenchymal mass was 29 × 20 issued in 2004 [1]. GISTs are known to consist of spindle and × 14 mm in size. Histopathological examination showed that the epithelioid cells. Among gastrointestinal mesenchymal tumors, specimens stained with hematoxylin and eosin-contained tumor GISTs are stained positively for c-KIT or CD34, and the diagnos- cells, which had elongated oval nuclei and eosinophilic and spin- tic rates of the tumors are <1% of all gastrointestinal tumors. dle-like reticulum, were arranged in fascicles without mitosis. Im- Gastrointestinal GISTs often appear in the stomach and small munohistochemical results were positive for c-KIT, S-100 protein, intestine. Esophageal GISTs are extremely rare, accounting for CD34 and vimentin, and the specimen was diagnosed as a only 1% of all GISTs [2, 3]. Esophagography, ultrasonography low-risk GIST (Fig. 4a and b). The patient took an uneventful and CT are used to diagnose GIST. Although the results of postoperative course, was allowed to remove the surgical drain FDG-PET have been reported to correlate with the degree of ma- after confirming the clinical status by esophagography on post- lignancy of GIST, the definitive diagnosisisdifficult to be per- operative day 9, and discharged on postoperative day 13. At 6 formed. On the other hand, FDG-PET is effective for evaluating years after surgery, the patient was found to be healthy without postoperative recurrence and the response of GIST to chemo- tumor recurrence. therapy [4]. For treating GIST, if the tumor is resectable, the par- tial resection of affected organ is the treatment of choice with considering the preservation of organ functions. In patients with unresectable tumors, recurrence or metastasis, chemo- DISCUSSION therapy with imatinib mesylate (Gleevec ) is recommended [1, 2]. Esophagectomy including partial resection, as a conven- In recent years, guidelines for managing GISTs are prepared by tional treatment for esophageal GIST, is more invasive than the National Comprehensive Cancer Network (NCCN) and T. Isaka et al. | 3 Figure 4: Histological specimens prepared from the removed GIST. The left and right microphotographs were taken at a magnification of ×40. (a) Hematoxylin and eosin- stained specimen showed spindle cells. (b) Immunohistochemical-stained specimen showed that the cells were positive for c-KIT. the resection of other parts of the gastrointestinal tract and re- REFERENCES markably reduces the quality of life of patients after surgery. Re- 1. Demetri GD, Benjamin RS, Blanke CD, Choi H, Corless C, cently, thoracoscopic enucleation has been used for treating DeMatteo RP, et al. NCCN task force report: optimal manage- small and low-risk GISTs confirmed histologically [5]. Previous ment of patients with gastrointestinal stromal tumor (GIST): case reports on esophageal GISTs have described only small expansion and update of NCCN clinical practice guidelines. numbers of patients, and long-term outcomes after enucleation J Natl Compr Canc Netw 2004;(Suppl 1):S1–26. have rarely been reported [6]. However, GISTs are difficult to be 2. Gupta P, Tewari M, Shukla HS. Gastrointestinal stromal tumor. diagnosed preoperatively, and esophageal submucosal tumors Surg Oncol 2008;17:129–38. such as leiomyoma are often initially diagnosed, followed by 3. Miettinen M, Lasota J. Gastrointestinal stromal tumors. the diagnosis of GIST on postoperative pathological examin- Gastroenterol Clin North Am 2013;42:399–415. ation [7]. The patient in this report was being followed up 6 4. Park JW, Cho CH, Jeong DS, Chae HD. Role of F-fluoro-2- years after surgery, and there was no evidence of recurrence. deoxyglucose positron emission tomography in gastric GIST: In patients with small tumors, diagnosis is frequently challen- predicting malignant potential pre-operatively. J Gastric ging. Minimally invasive thoracoscopic surgery could particularly Cancer 2011;11:173–9. be useful for the diagnosis and treatment of GIST appearing in 5. Koide N, Kishimoto K, Komatsu O, Yoshizawa A, Sugiyama A, the esophagus. This report described a patient who survived for Miyagawa S. Thoracoscopic enucleation of esophageal a long term without tumor recurrence after the thoracoscopic stromal tumor. Dis Esophagus 2004;17:104–8. enucleation of an esophageal GIST, considered a rare tumor. 6. Koyanagi K, Nakagawa M, Ozawa S, Nagase T, Seishima R, Being a lower aggressive procedure, the thoracoscopic enucle- Kanai T. Thoracoscopic enucleation for small-sized gastro- ation could allow patients to survive for a long term and their intestinal stromal tumor of the esophagus: report of two quality of life to be enhanced. cases. Esophagus 2010;7:219–24. 7. Nishimura K, Tanaka T, Tanaka Y, Matono S, Murata K, Naito Y, et al. Esophageal gastrointestinal stromal tumor- CONFLICT OF INTEREST STATEMENT incidence and prognosis after enucleation. Jpn J Gastroenterol None declared. Surg 2009;42:1551–6. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Long-term survival after thoracoscopic enucleation of a gastrointestinal stromal tumor arising from the esophagus

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Oxford University Press
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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2015.
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2042-8812
DOI
10.1093/jscr/rju155
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25656166
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Abstract

The goal of surgical treatment for gastrointestinal stromal tumor (GIST) is the complete resection of the tumor. A 62-year-old male had a clearly distinguishable mass having a smooth surface at the right side of the lower esophagus by computed tomography. Thoracoscopic resection of the tumor was performed. Immunohistochemical analysis showed that the tumor was positive for c-KIT and CD34 without mitosis, and diagnosed to be a low-risk GIST. At 6 years after surgery, the patient survived without recurrence. This study described the long-term surviving patient without the recurrence of tumor after the thoracoscopic resection of an esophageal GIST. INTRODUCTION Gastrointestinal stromal tumors (GISTs) in the esophagus are extremely rare. Reliable surgical therapy for GIST is the complete tumor resection. In principle, the resection of involved organ has been recommended for preserving physiological functions, and lymph-node dissection is unnecessary. This study described a patient who underwent the thoracoscopic enucleation of an esophageal GIST and survived without the recurrence of tumor for 6 years after surgery. CASE REPORT A 62-year-old male was found to have a well-demarcated mass with a smooth surface at the right side of the lower esophagus by chest computed tomography (CT) upon medical check-up (Fig. 1). Routine laboratory data and the serum levels of tumor markers were within normal limits. Positron emission tomography (PET) revealed the Figure 1: Chest CT of a 62-year-old male. The white arrow head indicates a clearly distinguishable mass having a smooth surface at the right side of the loweresophagus. accumulation of F-fluoro-2-deoxyglucose (FDG) at the tumor. Received: September 30, 2014. Revised: December 22, 2014. Accepted: December 29, 2014 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2015. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com 1 2 | Thoracoscopic resection of an esophageal GIST Figure 2: Endoscopic examination and endoscopic ultrasonography. (a) Tumor with a normally appearing mucosa was located 40 cm from the incisor teeth. (b) Hypoechoic submucosal tumor (asterisk) with annular localization, arising from the submucosal layer. The maximum standardized uptake value was 4.0, increasing to 4.8 in the late phase. Upper gastrointestinal endoscopy revealed no dis- tinct mucosal abnormalities or no evidence of tumors. Endoscopic ultrasonography showed a mass ∼2 cm in diameter arising in the third layer 40 cm from the incisor teeth (Fig. 2a and b). These find- ings diagnosed the lesion as an esophageal submucosal tumor with suspecting an esophageal leiomyoma or GIST. Being under general endotracheal double-lumen anesthesia with split-lung ventilation, the patient was allowed to lie on the healthy side (left) and posture lateral decubitus. A 6-cm long access incision was made at the pos- terior axillary line in the 10th intercostal space. Two 10.5-mm ports were made at the seventh intercostal space in the anterior axillary line and other at the same intercostal space in the posterior axillary line. After no adhesion in the thoracic cavity was observed, the pul- monary ligament was dissected and the lower lobe of the right lung was moved anteriorly. A tumefactive lesion was found in the lower esophagus. An incision was made in the mediastinal pleura, and Figure 3: Thoracoscopic view of the tumor. The image shows bulging through the the mass was confirmed at the outer longitudinal muscle of the right thoracic cavity. The black dash line and asterisk indicate the GIST; D, the lower esophagus. The tumor was easily removed from the sur- diaphragm; VD, the vertebral body. rounding tissue by a tumor enucleation procedure. No esophageal mucosa was injured intraoperatively. The operation time was 118 min, and the blood loss was 11 g (Fig. 3). Macroscopic examin- ation found that the removed white parenchymal mass was 29 × 20 issued in 2004 [1]. GISTs are known to consist of spindle and × 14 mm in size. Histopathological examination showed that the epithelioid cells. Among gastrointestinal mesenchymal tumors, specimens stained with hematoxylin and eosin-contained tumor GISTs are stained positively for c-KIT or CD34, and the diagnos- cells, which had elongated oval nuclei and eosinophilic and spin- tic rates of the tumors are <1% of all gastrointestinal tumors. dle-like reticulum, were arranged in fascicles without mitosis. Im- Gastrointestinal GISTs often appear in the stomach and small munohistochemical results were positive for c-KIT, S-100 protein, intestine. Esophageal GISTs are extremely rare, accounting for CD34 and vimentin, and the specimen was diagnosed as a only 1% of all GISTs [2, 3]. Esophagography, ultrasonography low-risk GIST (Fig. 4a and b). The patient took an uneventful and CT are used to diagnose GIST. Although the results of postoperative course, was allowed to remove the surgical drain FDG-PET have been reported to correlate with the degree of ma- after confirming the clinical status by esophagography on post- lignancy of GIST, the definitive diagnosisisdifficult to be per- operative day 9, and discharged on postoperative day 13. At 6 formed. On the other hand, FDG-PET is effective for evaluating years after surgery, the patient was found to be healthy without postoperative recurrence and the response of GIST to chemo- tumor recurrence. therapy [4]. For treating GIST, if the tumor is resectable, the par- tial resection of affected organ is the treatment of choice with considering the preservation of organ functions. In patients with unresectable tumors, recurrence or metastasis, chemo- DISCUSSION therapy with imatinib mesylate (Gleevec ) is recommended [1, 2]. Esophagectomy including partial resection, as a conven- In recent years, guidelines for managing GISTs are prepared by tional treatment for esophageal GIST, is more invasive than the National Comprehensive Cancer Network (NCCN) and T. Isaka et al. | 3 Figure 4: Histological specimens prepared from the removed GIST. The left and right microphotographs were taken at a magnification of ×40. (a) Hematoxylin and eosin- stained specimen showed spindle cells. (b) Immunohistochemical-stained specimen showed that the cells were positive for c-KIT. the resection of other parts of the gastrointestinal tract and re- REFERENCES markably reduces the quality of life of patients after surgery. Re- 1. Demetri GD, Benjamin RS, Blanke CD, Choi H, Corless C, cently, thoracoscopic enucleation has been used for treating DeMatteo RP, et al. NCCN task force report: optimal manage- small and low-risk GISTs confirmed histologically [5]. Previous ment of patients with gastrointestinal stromal tumor (GIST): case reports on esophageal GISTs have described only small expansion and update of NCCN clinical practice guidelines. numbers of patients, and long-term outcomes after enucleation J Natl Compr Canc Netw 2004;(Suppl 1):S1–26. have rarely been reported [6]. However, GISTs are difficult to be 2. Gupta P, Tewari M, Shukla HS. Gastrointestinal stromal tumor. diagnosed preoperatively, and esophageal submucosal tumors Surg Oncol 2008;17:129–38. such as leiomyoma are often initially diagnosed, followed by 3. Miettinen M, Lasota J. Gastrointestinal stromal tumors. the diagnosis of GIST on postoperative pathological examin- Gastroenterol Clin North Am 2013;42:399–415. ation [7]. The patient in this report was being followed up 6 4. Park JW, Cho CH, Jeong DS, Chae HD. Role of F-fluoro-2- years after surgery, and there was no evidence of recurrence. deoxyglucose positron emission tomography in gastric GIST: In patients with small tumors, diagnosis is frequently challen- predicting malignant potential pre-operatively. J Gastric ging. Minimally invasive thoracoscopic surgery could particularly Cancer 2011;11:173–9. be useful for the diagnosis and treatment of GIST appearing in 5. Koide N, Kishimoto K, Komatsu O, Yoshizawa A, Sugiyama A, the esophagus. This report described a patient who survived for Miyagawa S. Thoracoscopic enucleation of esophageal a long term without tumor recurrence after the thoracoscopic stromal tumor. Dis Esophagus 2004;17:104–8. enucleation of an esophageal GIST, considered a rare tumor. 6. Koyanagi K, Nakagawa M, Ozawa S, Nagase T, Seishima R, Being a lower aggressive procedure, the thoracoscopic enucle- Kanai T. Thoracoscopic enucleation for small-sized gastro- ation could allow patients to survive for a long term and their intestinal stromal tumor of the esophagus: report of two quality of life to be enhanced. cases. Esophagus 2010;7:219–24. 7. Nishimura K, Tanaka T, Tanaka Y, Matono S, Murata K, Naito Y, et al. Esophageal gastrointestinal stromal tumor- CONFLICT OF INTEREST STATEMENT incidence and prognosis after enucleation. Jpn J Gastroenterol None declared. Surg 2009;42:1551–6.

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Journal of Surgical Case ReportsOxford University Press

Published: Feb 5, 2015

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