Robot-assisted enucleation of large dumbbell-shaped esophageal schwannoma: a case report

Robot-assisted enucleation of large dumbbell-shaped esophageal schwannoma: a case report Background: Esophageal schwannomas are extremely rare, with few cases reported in the literature. Traditionally, resection of esophageal schwannoma is typically performed using thoracotomy or video-assisted thoracic surgery. However, large, irregular tumors may increase the surgical difficulties of complete enucleation and lead to potential mucosal damage. Moreover, a subtotal esophagectomy cannot be avoided in some conditions. Here, we report the first case of robot-assisted enucleation of a large dumbbell-shaped esophageal schwannoma. Case presentation: A 48-year-old woman presenting with a 1-year history of dysphagia was noted to have a homogeneous irregular mass measuring 70 mm in diameter and arising from the submucosal layer of the distal esophagus. A diagnosis of an esophageal submucosal tumor (SMT) was made, most likely leiomyoma. Robot-assisted thoracoscopic excision of the tumor was performed. The da Vinci Surgical System provided delicate dissection in the confined posterior mediastinal space, and the large dumbbell-shaped tumor was completely removed without damage to the mucosal integrity. The operative time was 108 min, and the blood loss was less than 20 ml. The pathology of the tumor was esophageal schwannoma. The patient experienced an unremarkable recovery and was discharged on the fifth day after operation. No symptoms or recurrence were present at the 50-month follow-up postoperatively. Conclusion: We present a rare case of large irregular esophageal schwannoma that was excised by robot-assisted surgery. A clear operative field and delicate dissections are critical points for the complete removal of this large esophageal SMT. We demonstrate that robotic treatment of large esophageal schwannoma is minimally invasive and can be successfully applied in such cases. Keywords: Esophageal schwannoma, Robot-assisted, Enucleation Background (VATS) [5–7]. Here, we report the first case of a large Esophageal schwannomas are extremely rare and are the irregular esophageal schwannoma that was successfully least common esophageal submucosal tumors (SMTs), removed via robot-assisted thoracoscopic surgery (RATS). with less than 30 reported cases in the literature [1]. The aim of this study is to describe the robot-assisted Preoperative differentiation of esophageal schwannomas surgical technique as well as the clinical and pathological from other SMTs is difficult, as there are no distinguish- features of this unusual tumor. ing characteristics regarding either symptoms or preoperative imaging tests [2]. A definitive diagnosis is Case presentation mostly established by pathological examinations after A 48-year-old woman presented to the gastroenterologist removal of the lesion [3, 4]. Surgical resection is the at a local hospital with a 1-year history of dysphagia, main treatment for esophageal schwannomas, usually via which had been progressively worsening over the prece- thoracotomy or video-assisted thoracoscopic surgery ding two months. During endoscopy, a bulging tumor 70 mm in length with an intact overlying mucosa was observed in the esophagus 30 cm away from the incisor * Correspondence: lihecheng2000@hotmail.com (Fig. 1a). Endoscopic ultrasonography (EUS) demonstrated Department of Thoracic Surgery, Ruijin Hospital, Shanghai Jiaotong University School of Medicine, 197 Ruijin 2nd Road, Shanghai 200025, China a hypoechoic and homogeneous mass in the submucosal © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Zhang et al. BMC Surgery (2018) 18:36 Page 2 of 5 Fig. 1 Preoperative imaging of the large esophageal SMT. (a) Upper endoscopy revealed a submucosal tumor 70 mm in length that was 30 cm from the incisor with an intact overlying mucosa. (b) CT scan of the chest revealed a 69 × 36 mm homogeneous mass in the distal esophageal wall (arrow) layer. Subsequently, a chest computed tomography (CT) the assistant surgeon on the patient’s left side. The hook or scan revealed a homogeneous irregular mass arising from Maryland was manipulated by the first arm of the robot. the posterior wall of the distal esophageal wall that was The Cadiere forceps was manipulated by the second arm. 69 × 36 mm in size and compressed the esophagus and The right lung was retracted laterally, exposing the esopha- trachea (Fig. 1b). Physical examinations, biochemical tests gus. By incising the mediastinal pleura, a large bulge was and cardiopulmonary function were normal. This patient clearly visualized in the distalesophagus in the confined had no medical or family history and was referred to our posterior mediastinal space (Fig. 3a). A longitudinal department for further treatment. Surgical evaluations myotomy was performed to expose the mass, in which a re- were discussed preoperatively. Based on the current traction suture was placed by 3–0 Vicryl (Ethicon US, LLC, imaging data, a diagnosis of esophageal SMT was made, Cincinnati, OH) and held by an assistant. The lesion was most likely leiomyoma. The patient was not recom- mended further preoperative endoscopic biopsy, which could probably result in mucosal adhesion to the tumor and a subsequently increased risk of mucosal injury during surgery. Taking into consideration the precarious location and large size of the tumor, a decision of robot-assisted resection surgery via the right transthoracic approach was made. The plan was to completely excise the tumor and maintain the integrity of the esophageal mucosa with a low threshold for conversion. Additionally, an esophagectomy with intrathoracic gastroesophagos- tomy was prepared if the tumor could not be removed. The surgical procedure was performed using a da Vinci Surgical System (Model S; Intuitive Surgical, Inc., Sunny- vale, CA, USA). After induction of general anesthesia and placement of a double-lumen endotracheal tube, the patient was placed in the left lateral decubitus position. A 12-mm camera port was inserted at the 10th intercostal space (ICS) at the midaxillary line. Under direct vision, three additional trocars were inserted under thoracoscopic guidance as fol- lows: an 8-mm port in the 7th ICS at the anterior axillary line for the first robotic arm, an 8-mm port in the 10th ICS at the posterior axillary line for the second robotic arm, and finally, a 12-mm auxiliary port in the 9th intercostal space immediately between the camera port and the first robotic port (Fig. 2). We used CO insufflation with 8– Fig. 2 Trocar placement for robot-assisted enucleation of the large 10 mmHg pressure. After all the trocars were positioned, esophageal schwannoma the robot was brought in from the head of the patient with Zhang et al. BMC Surgery (2018) 18:36 Page 3 of 5 Fig. 3 Robot-assisted enucleation of a large esophageal schwannoma. (a) By incising the mediastinal pleura, the large tumor was clearly visualized in the distal part of the esophagus. (b) The lesion was separated from the surrounding muscle using a combination of sharp and blunt dissection. (c) The split muscular layer and mediastinal pleura were loosely reapproximated with 2–0 Vicryl sutures. (d) The integrity of the mucosa was confirmed by simultaneous intra-operative upper endoscopy separated from the surrounding muscle and mucosa using liquid diet with good tolerance. She was discharged on a combination of sharp and blunt dissection under the fifth day after operation. Histopathological examin- three-dimensional vision and wrist-like movement of the ation revealed compact bundles of spindle cells (Fig. 4b). robotic instruments followed by enucleation (Fig. 3b). The Immunohistochemical staining was positive for S-100 split muscular layer and mediastinal pleura were loosely protein (Fig. 4c). The pathologic diagnosis of the tumor reapproximated with 2–0 Vicryl sutures (Fig. 3c). The was a benign esophageal schwannoma. The patient integrity of the mucosa was confirmed by air insufflation of remained asymptomatic and exhibited no evidence of the esophagus and upper endoscopic inspection (Fig. 3d). recurrence at the 50-month follow-up postoperatively. The lesion was removed from the thorax in a specimen retrieval bag. Grossly, the 70 × 60 × 40 mm tumor was Discussion and conclusions encapsulated (Fig. 4a). A 32-Fr chest tube was placed into Esophageal SMTs represent less than 1% of all esopha- one of the camera port sites under direct vision. The total geal neoplasms [8]. Of these lesions, leiomyomas are the operative time was 108 min, and the blood loss was less most common, accounting for 70–80% [9]. Esophageal than 20 ml. schwannomas, which arise from Schwann cells of the The patient was well postoperatively. On the third day, neural plexus within the esophageal wall, are the least a postoperative gastrograffin swallow demonstrated no common esophageal SMTs [10]. Esophageal schwannomas leaks or stricture. At this time, the patient started a occur frequently in middle-aged women in the proximal Fig. 4 (a) The specimen exhibited a well-encapsulated and dumbbell-shaped lesion that measured 70 × 60 × 40 mm. (b) Histopathological examination revealed compact bundles of spindle cells (asterisk) (Hematoxylin and eosinstain, original magnification, × 200). (c) Immunohistochemical staining showed positivity for S-100 protein (asterisk) (× 100) Zhang et al. BMC Surgery (2018) 18:36 Page 4 of 5 esophagus, with lesion dimensions varying from 1 cm to and location. The total enucleation of large tumors without 15 cm [1]. Similar to other esophageal SMTs, including damage to mucosal integrity is always a challenge due to leiomyomas and gastrointestinal stromal tumors (GIST), the limitations of the two-dimensional vision and range of schwannomas are often asymptomatic. If symptomatic, motion of the conventional thoracoscopic instruments. the most common presenting symptoms are dysphagia Watanabe et al. reported the difficulty of enucleation of an and chest discomfort [11]. Confirmation of diagnosis esophageal schwannoma larger than 5 cm using the VATS requires pathologic examination with further immuno- approach and the need to convert from enucleation to sub- staining studies after surgical resection. Tumor cells are total esophagectomy [7]. positive for S-100 protein but negative for smooth muscle Recently, RATS using the da Vinci Surgical System has markers, such as actin and desmin, which are positive in provided improved visualization and dexterity in esopha- leiomyoma, and for CD34 and CD117, which are charac- geal procedures. In our department, we began to perform teristically positive in GIST [2]. robot-assisted esophageal procedures on both malignant The main treatment for esophageal schwannomas is and benign tumors in May 2015. In the present case, the surgical resection [12]. Surgical indications include the tumor was located within the esophageal wall in the con- presence of symptoms, evidence of an increase in tumor fined posterior mediastinum and was 70 mm in diameter. size, and the need to confirm the pathologic diagnosis [2]. Furthermore, this submucosal mass was irregular with a Esophageal schwannomas are commonly benign, with dumbbell-like shape, which increased the difficulty of only a few reports of malignant cases [13, 14]. Tumor obtaining complete enucleation. A decision of transtho- enucleation, as opposed to complete curative resection, is racic tumor enucleation using the da Vinci Surgical Sys- generally sufficient for benign schwannomas [3–5]. The tem was made. The robotic approach offers advantages conventional treatment for esophageal schwannomas is compared with conventional thoracoscopic systems, transthoracic enucleation through thoracotomy. Currently, including wrist-like movement of the instruments, VATS is more popular because it is less painful and offers a three-dimensional vision and ergonomic comfort for the shorter recovery time than thoracotomy [15]. There are 8 surgeon. These features facilitated the combination of reported cases of esophageal schwannoma resection by sharp and blunt dissection in the confined space and VATS in the literature (Table 1). However, the possibility of subsequently offered the possibility of complete removal thoracoscopic enucleation may be limited by the tumor size of the tumor without interruption of the capsule and the Table 1 Literature review of esophageal schwannoma resections by minimally invasive surgery Case Author Year Age Location Size Symptoms Surgical Management Conversion Operating Complications LOS (years)/ (mm) Approach time (min) (d) Sex 1 Chen et 2006 73/W Ut 45 × Cough, VATS Enucleation None NA None NA al. [6] 50 × 70 dyspnea, dysphagia 2 Mizuguchi 2008 20/W Ut-Mt 80 × Dyspnea VATS Enucleation None NA None 15 et al. [16] 75 × 60 3 Toyama 2008 37/W Ut 28 × None VATS Enucleation None NA None 4 et al. [17] 24 × 19 4 Makino 2013 72/M Ut 22 × None VATS Enucleation None NA None NA et al. [18] 34 × 29 5 Shichinohe 2014 61/W Lt 40 × Dysphagia VATS Enucleation None 174 None 8 et al. [19] 30 × 45 6 Chen et al. 2016 46/M Mt 30 × 20 Discomfort VATS Enucleation None NA None 5 [20] × 17/30 × during 18 × 15 swallowing 7 Watanabe 2016 39/W Ut 39 × Difficulty VATS Subtotal Yes NA None NA et al. [7] 28 × 56 swallowing, esophagectomy (enucleation to epigastric esophagectomy) pain 8 Onodera 2017 47/W Mt-Lt 60 Dysphagia VATS Enucleation None 498 None 9 et al. [21] 9 Our case 2018 48/W Lt 70 × Dysphagia RATS Enucleation None 108 None 5 60 × 40 W, woman; M, man; Ut, upper thoracic esophagus; Mt, middle thoracic esophagus; Lt, lower thoracic esophagus; VATS, video-assisted thoracoscopic surgery; RATS, robot-assisted thoracoscopic surgery; LOS, length of stay Zhang et al. BMC Surgery (2018) 18:36 Page 5 of 5 surrounding esophageal mucosa. The integrity of the mu- Received: 2 May 2018 Accepted: 25 May 2018 cosa was ensured by the precise dissection with robotic surgery and simultaneous intraoperative upper endoscopy. References Moreover, the suture and knot for the repair of the myo- 1. Park BJ, Carrasquillo J, Bains MS, Flores RM. Giant benign esophageal schwannoma requiring esophagectomy. Ann Thorac Surg. 2006;82:340–2. tomy could be easily performed by the wristed robotic in- 2. Ha C, Regan J, Cetindag IB, Ali A, Mellinger JD. Benign esophageal tumors. struments, which prevented mucosal bulging and possible Surg Clin North Am. 2015;95:491–514. formation of a diverticulum after the enucleation of the 3. Kobayashi N, Kikuchi S, Shimao H, Hiki Y, Kakita A, Mitomi H, et al. Benign esophageal schwannoma: report of a case. Surg Today. 2000;30:526–9. large esophageal SMT. 4. Kitada M, Matsuda Y, Hayashi S, Ishibashi K, Oikawa K, Miyokawa N. To the best of our knowledge, this is the first report of Esophageal schwannoma: a case report. World J Surg Oncol. 2013;11:253. robot-assisted enucleation of a large esophageal schwan- 5. Tokunaga T, Takeda S, Sumimura J, Maeda H. Esophageal schwannoma: report of a case. Surg Today. 2007;37:500–2. noma. A clear operative field and delicate dissections are 6. Chen HC, Huang HJ, Wu CY, Lin TS, Fang HY. Esophageal schwannoma with key points. This illustrative case demonstrates the utility of tracheal compression. Thorac Cardiovasc Surg. 2006;54:555–8. robotic surgery in the removal of a large irregular esophageal 7. Watanabe T, Miyazaki T, Saito H, Yoshida T, Kumakura Y, Honjyo H, et al. Resection of an esophageal schwannoma with thoracoscopic surgery: a SMT with respect to its safety and minimal invasiveness. case report. Surg Case Rep. 2016;2:127. 8. Postlethwait RW, Musser AW. Changes in the esophagus in 1,000 autopsy specimens. J Thorac Cardiovasc Surg. 1974;68:953–6. Abbreviations 9. Seremetis MG, Lyons WS, deGuzman VC, Peabody JW Jr. Leiomyomata of SMT: Submucosal tumor; VATS: Video-assisted thoracoscopic surgery; the esophagus. An analysis of 838 cases. Cancer 1976;38:2166–2177. RATS: Robot-assisted thoracoscopic surgery; EUS: Endoscopic 10. Kwon MS, Lee SS, Ahn GH. Schwannomas of the gastrointestinal tract: ultrasonography; CT: Computed tomography; ICS: Intercostal space; clinicopathological features of 12 cases including a case of esophageal GIST: Gastrointestinal stromal tumor; LOS: Length of stay tumor compared with those of gastrointestinal stromal tumors and leiomyomas of the gastrointestinal tract. Pathol Res Pract. 2002;198:605–13. Acknowledgements 11. Kassis ES, Bansal S, Perrino C, Walker JP, Hitchcock C, Ross P Jr, et al. We thank the subjects for their participation. We appreciate very much for Dr. Giant asymptomatic primary esophageal schwannoma. Ann Thorac Chao-fu Wang’s kindly help with the pathologic pictures of the esophageal Surg. 2012;93:e81–3. schwannoma. We also appreciate Dr. Shi-Dan Cheng’shelp withthe 12. Saito R, Kitamura M, Suzuki H, Ogawa J, Sageshima M, et al. Esophageal introperative upper endoscopy. schwannoma. Ann Thorac Surg. 2000;69:1947–9. 13. Mishra B, Madhusudhan KS, Kilambi R, Das P, Pal S, Srivastava DN. Malignant schwannoma of the esophagus: a rare case report. Korean J Thorac Competing interests. Cardiovasc Surg. 2016;49:63–6. The authors declare that they have no competing interests. 14. Wang S, Zheng J, Ruan Z, Huang H, Yang Z, Zheng J. Long-term survival in a rare case of malignant esophageal schwannoma cured by surgical excision. Ann Thorac Surg. 2011;92:357–8. Funding 15. Luh SP, Liu HP. Video-assisted thoracic surgery–the past, present status and This work was supported by grants from the Shanghai Municipal Education the future. J Zhejiang Univ Sci. 2006;B7:118–28. Commission-Gaofeng Clinical Medicine Grant Support (20172005) and 16. Mizuguchi S, Inoue K, Imagawa A, Kitano Y, Kameyama M, Ueda H, et al. Shanghai Jiao Tong University Cooperation Grant of Medicine, Science, Benign esophageal schwannoma compressing the trachea in pregnancy. Engineering (YG2015QN39). The funders had no role in the study design, Ann Thorac Surg. 2008;85:660–2. data collection and analysis, decision to publish, or preparation of the 17. Toyama E, Nagai Y, Baba Y, Baba Y, Yoshida N, Hayashi N, et al. A case of manuscript. thoracoscopically resected benign esophageal schwannoma with high uptake on FDG-PET. Esophagus. 2008;5:167–70. Availability of data and materials. 18. Makino T, Yamasaki M, Takeno A, Kurokawa Y, Miyata H, Takiguchi S, et al. All data generated or analyzed during this study are included in this Thoracoscopic enucleation of esophageal schwannoma exhibiting (18) F- published article. fluorodeoxyglucose uptake on positron emission tomography. Dis Esophagus. 2013;26:331–2. 19. Shichinohe T, Kato K, Ebihara Y, Kurashima Y, Tsuchikawa T, Matsumoto J, et Authors’ contributions. al. Thoracoscopic enucleation of esophageal submucosal tumor by prone HCL designed the study and supervised the study. YJZ, YH and JX collected position under artificial pneumothorax by CO2 insufflation. Surg Laparosc the data. YJZ, YH, JX and HCL performed the surgery. JX and YH did the Endosc Percutan Tech. 2014;24:e55–8. follow-up. YJZ drafted the manuscript. All of the co-authors have revised this 20. Chen X, Li Y, Liu X, Fu H, Sun H, Zhang R, et al. A report of three cases of manuscript and support this submission. All authors read and approved the surgical removal of esophageal schwannomas. J Thorac Dis. 2016;8:E353–7. final manuscript. 21. Onodera Y, Nakano T, Takeyama D, Maruyama S, Taniyama Y, Sakurai T, et al. Combined thoracoscopic and endoscopic surgery for a large esophageal schwannoma. World J Gastroenterol. 2017;23:8256–60. Consent for publication. Written informed consent was obtained from the patient for the publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal. Ethics approval and consent to participate. Not applicable. Publisher’sNote Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png BMC Surgery Springer Journals

Robot-assisted enucleation of large dumbbell-shaped esophageal schwannoma: a case report

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Abstract

Background: Esophageal schwannomas are extremely rare, with few cases reported in the literature. Traditionally, resection of esophageal schwannoma is typically performed using thoracotomy or video-assisted thoracic surgery. However, large, irregular tumors may increase the surgical difficulties of complete enucleation and lead to potential mucosal damage. Moreover, a subtotal esophagectomy cannot be avoided in some conditions. Here, we report the first case of robot-assisted enucleation of a large dumbbell-shaped esophageal schwannoma. Case presentation: A 48-year-old woman presenting with a 1-year history of dysphagia was noted to have a homogeneous irregular mass measuring 70 mm in diameter and arising from the submucosal layer of the distal esophagus. A diagnosis of an esophageal submucosal tumor (SMT) was made, most likely leiomyoma. Robot-assisted thoracoscopic excision of the tumor was performed. The da Vinci Surgical System provided delicate dissection in the confined posterior mediastinal space, and the large dumbbell-shaped tumor was completely removed without damage to the mucosal integrity. The operative time was 108 min, and the blood loss was less than 20 ml. The pathology of the tumor was esophageal schwannoma. The patient experienced an unremarkable recovery and was discharged on the fifth day after operation. No symptoms or recurrence were present at the 50-month follow-up postoperatively. Conclusion: We present a rare case of large irregular esophageal schwannoma that was excised by robot-assisted surgery. A clear operative field and delicate dissections are critical points for the complete removal of this large esophageal SMT. We demonstrate that robotic treatment of large esophageal schwannoma is minimally invasive and can be successfully applied in such cases. Keywords: Esophageal schwannoma, Robot-assisted, Enucleation Background (VATS) [5–7]. Here, we report the first case of a large Esophageal schwannomas are extremely rare and are the irregular esophageal schwannoma that was successfully least common esophageal submucosal tumors (SMTs), removed via robot-assisted thoracoscopic surgery (RATS). with less than 30 reported cases in the literature [1]. The aim of this study is to describe the robot-assisted Preoperative differentiation of esophageal schwannomas surgical technique as well as the clinical and pathological from other SMTs is difficult, as there are no distinguish- features of this unusual tumor. ing characteristics regarding either symptoms or preoperative imaging tests [2]. A definitive diagnosis is Case presentation mostly established by pathological examinations after A 48-year-old woman presented to the gastroenterologist removal of the lesion [3, 4]. Surgical resection is the at a local hospital with a 1-year history of dysphagia, main treatment for esophageal schwannomas, usually via which had been progressively worsening over the prece- thoracotomy or video-assisted thoracoscopic surgery ding two months. During endoscopy, a bulging tumor 70 mm in length with an intact overlying mucosa was observed in the esophagus 30 cm away from the incisor * Correspondence: lihecheng2000@hotmail.com (Fig. 1a). Endoscopic ultrasonography (EUS) demonstrated Department of Thoracic Surgery, Ruijin Hospital, Shanghai Jiaotong University School of Medicine, 197 Ruijin 2nd Road, Shanghai 200025, China a hypoechoic and homogeneous mass in the submucosal © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Zhang et al. BMC Surgery (2018) 18:36 Page 2 of 5 Fig. 1 Preoperative imaging of the large esophageal SMT. (a) Upper endoscopy revealed a submucosal tumor 70 mm in length that was 30 cm from the incisor with an intact overlying mucosa. (b) CT scan of the chest revealed a 69 × 36 mm homogeneous mass in the distal esophageal wall (arrow) layer. Subsequently, a chest computed tomography (CT) the assistant surgeon on the patient’s left side. The hook or scan revealed a homogeneous irregular mass arising from Maryland was manipulated by the first arm of the robot. the posterior wall of the distal esophageal wall that was The Cadiere forceps was manipulated by the second arm. 69 × 36 mm in size and compressed the esophagus and The right lung was retracted laterally, exposing the esopha- trachea (Fig. 1b). Physical examinations, biochemical tests gus. By incising the mediastinal pleura, a large bulge was and cardiopulmonary function were normal. This patient clearly visualized in the distalesophagus in the confined had no medical or family history and was referred to our posterior mediastinal space (Fig. 3a). A longitudinal department for further treatment. Surgical evaluations myotomy was performed to expose the mass, in which a re- were discussed preoperatively. Based on the current traction suture was placed by 3–0 Vicryl (Ethicon US, LLC, imaging data, a diagnosis of esophageal SMT was made, Cincinnati, OH) and held by an assistant. The lesion was most likely leiomyoma. The patient was not recom- mended further preoperative endoscopic biopsy, which could probably result in mucosal adhesion to the tumor and a subsequently increased risk of mucosal injury during surgery. Taking into consideration the precarious location and large size of the tumor, a decision of robot-assisted resection surgery via the right transthoracic approach was made. The plan was to completely excise the tumor and maintain the integrity of the esophageal mucosa with a low threshold for conversion. Additionally, an esophagectomy with intrathoracic gastroesophagos- tomy was prepared if the tumor could not be removed. The surgical procedure was performed using a da Vinci Surgical System (Model S; Intuitive Surgical, Inc., Sunny- vale, CA, USA). After induction of general anesthesia and placement of a double-lumen endotracheal tube, the patient was placed in the left lateral decubitus position. A 12-mm camera port was inserted at the 10th intercostal space (ICS) at the midaxillary line. Under direct vision, three additional trocars were inserted under thoracoscopic guidance as fol- lows: an 8-mm port in the 7th ICS at the anterior axillary line for the first robotic arm, an 8-mm port in the 10th ICS at the posterior axillary line for the second robotic arm, and finally, a 12-mm auxiliary port in the 9th intercostal space immediately between the camera port and the first robotic port (Fig. 2). We used CO insufflation with 8– Fig. 2 Trocar placement for robot-assisted enucleation of the large 10 mmHg pressure. After all the trocars were positioned, esophageal schwannoma the robot was brought in from the head of the patient with Zhang et al. BMC Surgery (2018) 18:36 Page 3 of 5 Fig. 3 Robot-assisted enucleation of a large esophageal schwannoma. (a) By incising the mediastinal pleura, the large tumor was clearly visualized in the distal part of the esophagus. (b) The lesion was separated from the surrounding muscle using a combination of sharp and blunt dissection. (c) The split muscular layer and mediastinal pleura were loosely reapproximated with 2–0 Vicryl sutures. (d) The integrity of the mucosa was confirmed by simultaneous intra-operative upper endoscopy separated from the surrounding muscle and mucosa using liquid diet with good tolerance. She was discharged on a combination of sharp and blunt dissection under the fifth day after operation. Histopathological examin- three-dimensional vision and wrist-like movement of the ation revealed compact bundles of spindle cells (Fig. 4b). robotic instruments followed by enucleation (Fig. 3b). The Immunohistochemical staining was positive for S-100 split muscular layer and mediastinal pleura were loosely protein (Fig. 4c). The pathologic diagnosis of the tumor reapproximated with 2–0 Vicryl sutures (Fig. 3c). The was a benign esophageal schwannoma. The patient integrity of the mucosa was confirmed by air insufflation of remained asymptomatic and exhibited no evidence of the esophagus and upper endoscopic inspection (Fig. 3d). recurrence at the 50-month follow-up postoperatively. The lesion was removed from the thorax in a specimen retrieval bag. Grossly, the 70 × 60 × 40 mm tumor was Discussion and conclusions encapsulated (Fig. 4a). A 32-Fr chest tube was placed into Esophageal SMTs represent less than 1% of all esopha- one of the camera port sites under direct vision. The total geal neoplasms [8]. Of these lesions, leiomyomas are the operative time was 108 min, and the blood loss was less most common, accounting for 70–80% [9]. Esophageal than 20 ml. schwannomas, which arise from Schwann cells of the The patient was well postoperatively. On the third day, neural plexus within the esophageal wall, are the least a postoperative gastrograffin swallow demonstrated no common esophageal SMTs [10]. Esophageal schwannomas leaks or stricture. At this time, the patient started a occur frequently in middle-aged women in the proximal Fig. 4 (a) The specimen exhibited a well-encapsulated and dumbbell-shaped lesion that measured 70 × 60 × 40 mm. (b) Histopathological examination revealed compact bundles of spindle cells (asterisk) (Hematoxylin and eosinstain, original magnification, × 200). (c) Immunohistochemical staining showed positivity for S-100 protein (asterisk) (× 100) Zhang et al. BMC Surgery (2018) 18:36 Page 4 of 5 esophagus, with lesion dimensions varying from 1 cm to and location. The total enucleation of large tumors without 15 cm [1]. Similar to other esophageal SMTs, including damage to mucosal integrity is always a challenge due to leiomyomas and gastrointestinal stromal tumors (GIST), the limitations of the two-dimensional vision and range of schwannomas are often asymptomatic. If symptomatic, motion of the conventional thoracoscopic instruments. the most common presenting symptoms are dysphagia Watanabe et al. reported the difficulty of enucleation of an and chest discomfort [11]. Confirmation of diagnosis esophageal schwannoma larger than 5 cm using the VATS requires pathologic examination with further immuno- approach and the need to convert from enucleation to sub- staining studies after surgical resection. Tumor cells are total esophagectomy [7]. positive for S-100 protein but negative for smooth muscle Recently, RATS using the da Vinci Surgical System has markers, such as actin and desmin, which are positive in provided improved visualization and dexterity in esopha- leiomyoma, and for CD34 and CD117, which are charac- geal procedures. In our department, we began to perform teristically positive in GIST [2]. robot-assisted esophageal procedures on both malignant The main treatment for esophageal schwannomas is and benign tumors in May 2015. In the present case, the surgical resection [12]. Surgical indications include the tumor was located within the esophageal wall in the con- presence of symptoms, evidence of an increase in tumor fined posterior mediastinum and was 70 mm in diameter. size, and the need to confirm the pathologic diagnosis [2]. Furthermore, this submucosal mass was irregular with a Esophageal schwannomas are commonly benign, with dumbbell-like shape, which increased the difficulty of only a few reports of malignant cases [13, 14]. Tumor obtaining complete enucleation. A decision of transtho- enucleation, as opposed to complete curative resection, is racic tumor enucleation using the da Vinci Surgical Sys- generally sufficient for benign schwannomas [3–5]. The tem was made. The robotic approach offers advantages conventional treatment for esophageal schwannomas is compared with conventional thoracoscopic systems, transthoracic enucleation through thoracotomy. Currently, including wrist-like movement of the instruments, VATS is more popular because it is less painful and offers a three-dimensional vision and ergonomic comfort for the shorter recovery time than thoracotomy [15]. There are 8 surgeon. These features facilitated the combination of reported cases of esophageal schwannoma resection by sharp and blunt dissection in the confined space and VATS in the literature (Table 1). However, the possibility of subsequently offered the possibility of complete removal thoracoscopic enucleation may be limited by the tumor size of the tumor without interruption of the capsule and the Table 1 Literature review of esophageal schwannoma resections by minimally invasive surgery Case Author Year Age Location Size Symptoms Surgical Management Conversion Operating Complications LOS (years)/ (mm) Approach time (min) (d) Sex 1 Chen et 2006 73/W Ut 45 × Cough, VATS Enucleation None NA None NA al. [6] 50 × 70 dyspnea, dysphagia 2 Mizuguchi 2008 20/W Ut-Mt 80 × Dyspnea VATS Enucleation None NA None 15 et al. [16] 75 × 60 3 Toyama 2008 37/W Ut 28 × None VATS Enucleation None NA None 4 et al. [17] 24 × 19 4 Makino 2013 72/M Ut 22 × None VATS Enucleation None NA None NA et al. [18] 34 × 29 5 Shichinohe 2014 61/W Lt 40 × Dysphagia VATS Enucleation None 174 None 8 et al. [19] 30 × 45 6 Chen et al. 2016 46/M Mt 30 × 20 Discomfort VATS Enucleation None NA None 5 [20] × 17/30 × during 18 × 15 swallowing 7 Watanabe 2016 39/W Ut 39 × Difficulty VATS Subtotal Yes NA None NA et al. [7] 28 × 56 swallowing, esophagectomy (enucleation to epigastric esophagectomy) pain 8 Onodera 2017 47/W Mt-Lt 60 Dysphagia VATS Enucleation None 498 None 9 et al. [21] 9 Our case 2018 48/W Lt 70 × Dysphagia RATS Enucleation None 108 None 5 60 × 40 W, woman; M, man; Ut, upper thoracic esophagus; Mt, middle thoracic esophagus; Lt, lower thoracic esophagus; VATS, video-assisted thoracoscopic surgery; RATS, robot-assisted thoracoscopic surgery; LOS, length of stay Zhang et al. BMC Surgery (2018) 18:36 Page 5 of 5 surrounding esophageal mucosa. The integrity of the mu- Received: 2 May 2018 Accepted: 25 May 2018 cosa was ensured by the precise dissection with robotic surgery and simultaneous intraoperative upper endoscopy. References Moreover, the suture and knot for the repair of the myo- 1. Park BJ, Carrasquillo J, Bains MS, Flores RM. Giant benign esophageal schwannoma requiring esophagectomy. Ann Thorac Surg. 2006;82:340–2. tomy could be easily performed by the wristed robotic in- 2. Ha C, Regan J, Cetindag IB, Ali A, Mellinger JD. Benign esophageal tumors. struments, which prevented mucosal bulging and possible Surg Clin North Am. 2015;95:491–514. formation of a diverticulum after the enucleation of the 3. Kobayashi N, Kikuchi S, Shimao H, Hiki Y, Kakita A, Mitomi H, et al. Benign esophageal schwannoma: report of a case. Surg Today. 2000;30:526–9. large esophageal SMT. 4. Kitada M, Matsuda Y, Hayashi S, Ishibashi K, Oikawa K, Miyokawa N. To the best of our knowledge, this is the first report of Esophageal schwannoma: a case report. World J Surg Oncol. 2013;11:253. robot-assisted enucleation of a large esophageal schwan- 5. Tokunaga T, Takeda S, Sumimura J, Maeda H. Esophageal schwannoma: report of a case. Surg Today. 2007;37:500–2. noma. A clear operative field and delicate dissections are 6. Chen HC, Huang HJ, Wu CY, Lin TS, Fang HY. Esophageal schwannoma with key points. This illustrative case demonstrates the utility of tracheal compression. Thorac Cardiovasc Surg. 2006;54:555–8. robotic surgery in the removal of a large irregular esophageal 7. Watanabe T, Miyazaki T, Saito H, Yoshida T, Kumakura Y, Honjyo H, et al. Resection of an esophageal schwannoma with thoracoscopic surgery: a SMT with respect to its safety and minimal invasiveness. case report. Surg Case Rep. 2016;2:127. 8. Postlethwait RW, Musser AW. Changes in the esophagus in 1,000 autopsy specimens. J Thorac Cardiovasc Surg. 1974;68:953–6. Abbreviations 9. Seremetis MG, Lyons WS, deGuzman VC, Peabody JW Jr. Leiomyomata of SMT: Submucosal tumor; VATS: Video-assisted thoracoscopic surgery; the esophagus. An analysis of 838 cases. Cancer 1976;38:2166–2177. RATS: Robot-assisted thoracoscopic surgery; EUS: Endoscopic 10. Kwon MS, Lee SS, Ahn GH. Schwannomas of the gastrointestinal tract: ultrasonography; CT: Computed tomography; ICS: Intercostal space; clinicopathological features of 12 cases including a case of esophageal GIST: Gastrointestinal stromal tumor; LOS: Length of stay tumor compared with those of gastrointestinal stromal tumors and leiomyomas of the gastrointestinal tract. Pathol Res Pract. 2002;198:605–13. Acknowledgements 11. Kassis ES, Bansal S, Perrino C, Walker JP, Hitchcock C, Ross P Jr, et al. We thank the subjects for their participation. We appreciate very much for Dr. Giant asymptomatic primary esophageal schwannoma. Ann Thorac Chao-fu Wang’s kindly help with the pathologic pictures of the esophageal Surg. 2012;93:e81–3. schwannoma. We also appreciate Dr. Shi-Dan Cheng’shelp withthe 12. Saito R, Kitamura M, Suzuki H, Ogawa J, Sageshima M, et al. Esophageal introperative upper endoscopy. schwannoma. Ann Thorac Surg. 2000;69:1947–9. 13. Mishra B, Madhusudhan KS, Kilambi R, Das P, Pal S, Srivastava DN. Malignant schwannoma of the esophagus: a rare case report. Korean J Thorac Competing interests. Cardiovasc Surg. 2016;49:63–6. The authors declare that they have no competing interests. 14. Wang S, Zheng J, Ruan Z, Huang H, Yang Z, Zheng J. Long-term survival in a rare case of malignant esophageal schwannoma cured by surgical excision. Ann Thorac Surg. 2011;92:357–8. Funding 15. Luh SP, Liu HP. Video-assisted thoracic surgery–the past, present status and This work was supported by grants from the Shanghai Municipal Education the future. J Zhejiang Univ Sci. 2006;B7:118–28. Commission-Gaofeng Clinical Medicine Grant Support (20172005) and 16. Mizuguchi S, Inoue K, Imagawa A, Kitano Y, Kameyama M, Ueda H, et al. Shanghai Jiao Tong University Cooperation Grant of Medicine, Science, Benign esophageal schwannoma compressing the trachea in pregnancy. Engineering (YG2015QN39). The funders had no role in the study design, Ann Thorac Surg. 2008;85:660–2. data collection and analysis, decision to publish, or preparation of the 17. Toyama E, Nagai Y, Baba Y, Baba Y, Yoshida N, Hayashi N, et al. A case of manuscript. thoracoscopically resected benign esophageal schwannoma with high uptake on FDG-PET. Esophagus. 2008;5:167–70. Availability of data and materials. 18. Makino T, Yamasaki M, Takeno A, Kurokawa Y, Miyata H, Takiguchi S, et al. All data generated or analyzed during this study are included in this Thoracoscopic enucleation of esophageal schwannoma exhibiting (18) F- published article. fluorodeoxyglucose uptake on positron emission tomography. Dis Esophagus. 2013;26:331–2. 19. Shichinohe T, Kato K, Ebihara Y, Kurashima Y, Tsuchikawa T, Matsumoto J, et Authors’ contributions. al. Thoracoscopic enucleation of esophageal submucosal tumor by prone HCL designed the study and supervised the study. YJZ, YH and JX collected position under artificial pneumothorax by CO2 insufflation. Surg Laparosc the data. YJZ, YH, JX and HCL performed the surgery. JX and YH did the Endosc Percutan Tech. 2014;24:e55–8. follow-up. YJZ drafted the manuscript. All of the co-authors have revised this 20. Chen X, Li Y, Liu X, Fu H, Sun H, Zhang R, et al. A report of three cases of manuscript and support this submission. All authors read and approved the surgical removal of esophageal schwannomas. J Thorac Dis. 2016;8:E353–7. final manuscript. 21. Onodera Y, Nakano T, Takeyama D, Maruyama S, Taniyama Y, Sakurai T, et al. Combined thoracoscopic and endoscopic surgery for a large esophageal schwannoma. World J Gastroenterol. 2017;23:8256–60. Consent for publication. Written informed consent was obtained from the patient for the publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal. Ethics approval and consent to participate. Not applicable. Publisher’sNote Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Journal

BMC SurgerySpringer Journals

Published: Jun 5, 2018

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