Endoscopic Submucosal Tunnel Dissection for ...

Video Journal and Encyclopedia of GI Endoscopy (]]]]) ], ]]]–]]]

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Available online at www.sciencedirect.com

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CLINICAL CASE REPORTS

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Endoscopic Submucosal Tunnel Dissection for Esophageal Granular Cell Tumor Resection$, $$

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Francisco Baldaque-Silvaa,n, Margarida Marquesa, Joanne Lopesb, Guilherme Macedoa

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Gastroenterology Department, Centro Hospitalar de São João, Alameda Professor Hernani Monteiro, Porto, Portugal b Pathology Department, Centro Hospitalar de São João, Alameda Professor Hernani Monteiro, Q2 Porto, Portugal Received 11 January 2013; received in revised form 27 April 2013; accepted 22 May 2013

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KEYWORDS

Abstract

Endoscopic submucosal tunnel dissection; Granular cell tumor; Submucosal tumor; Esophagus; Video

Endoscopic submucosal tunnel dissection (ESTD) was recently described for the resection of upper gastrointestinal submucosal tumors, namely leiomyomas, GISTs and aberrant pancreas. Granular cell tumors (GCT) are usually benign, but should be removed when symptomatic, significantly increase in size or have atypical histological or ultrasonographic features. We aim to describe the role of ESTD for the resection of an esophageal GCT. A 51 year-old patient was referred to us due to the presence of an esophageal submucosal lesion with increased size in the follow-up. Deep biopsy specimens were positive for granular cell tumor. Suboptimal submucosal lifting precluded conventional endoscopic submucosal dissection (ESD). In this context an ESTD was performed. First, a submucosal tunnel was created starting 5 cm above the tumor. Afterwards, the GCT was carefully dissected from the overlying submucosa and muscularis propria using TT knife and IT knife2. The ESTD procedure was possible and en bloc resection achieved, being the 25 mm long lesion retrieved. The mucosal orifices were closed using conventional clips. The patient started oral diet 1 day after ESTD and was discharged at day 4 without any complications. In this first report of ESTD for esophageal GCT resection, this technique shown to be feasible, reliable and safe, enabling complete resection, even in this case with poor submucosal lifting. & 2013 The Authors. Published by Elsevier GmbH. All rights reserved.

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This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial-No Derivative Works License, which permits non-commercial use, distribution, and reproduction in any medium, provided the original author and source are credited. ☆☆ The terms of this license also apply to the corresponding video. n Q3 Corresponding author. Tel.: +351 936641601. E-mail addresses: [email protected] (F. Baldaque-Silva), [email protected] (M. Marques), [email protected] (J. Lopes), [email protected] (G. Macedo). 2212-0971/$ - see front matter & 2013 The Authors. Published by Elsevier GmbH. All rights reserved. http://dx.doi.org/10.1016/j.vjgien.2013.05.004 Please cite this article as: Baldaque-Silva F, et al. Endoscopic Submucosal Tunnel Dissection for Esophageal Granular Cell Tumor Resection. Video Journal and Encyclopedia of GI Endoscopy (2013), http://dx.doi.org/10.1016/j.vjgien.2013.05.004

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Video related to this article

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Video related to this article can be found online at http:// dx.doi.org/10.1016/j.vjgien.2013.05.004.

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pump inhibitor therapy. Esophageal submucosal lesion with increased size in the follow-up. Biopsy specimens positive for granular cell tumor. Endoscopic ultrasound shows a lesion restricted to the submucosal layer. Poor submucosal lifting precluded conventional ESD resection. Endoscopic submucosal tunnel dissection was performed. Patient started oral diet on day 1 and was discharged asymptomatic at day 4. Patient remained asymptomatic at the 2 and 6 months follow-up.

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Gastroscope GIF-H180 and GIF-HQ190 (Olympus Corp.,

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Spray catheter (PW-6P-1; Olympus). Interject sclerotherapy needle (1835, Boston Scientific,

Materials Hamburg, Germany).

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Natick, MA, USA). Transparent cap (DH-29CR, Fujinon, Omiya, Japan). Saline solution with diluted epinephrine (1:200,000) and methylene blue. Triangular tip knife (TT knife, KD-640L; Olympus). Insulation tipped IT-knife2 (KD-611L; Olympus). Coagulation forceps (FD-410LR, Coagrasper; Olympus). Quick clip 2 (HX-201LR-135.A, Olympus). Carbon dioxide gas for insuflation. Electrosurgery unit (Erbe Vio 300S; Erbe Elektromedizin, Tubingen, Germany).

Endoscopic procedure

Intravenous metronidazole and ciprofloxacin are given

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Case report

51 year-old male patient. History of erosive esophagitis non-responsive to proton

Delineation and dissection of the submucosal side of the

before the procedure and maintained in the first day. Intravenous proton pump inhibitor (PPI) is started before the procedure and changed to oral PPI after resumption of oral diet. The esophagus is rinsed with saline and chlorhexidine using a catheter spray. Mucosal entry is created by mucosal lifting 5 cm above the submucosal lesion, using the sclerotherapy needle and a saline solution with diluted adrenalin and methylene blue. Subsequent longitudinal incision is made using TT knife in endocut mode effect 2. Using continuous endoscopeknife traction a 5 cm long incision is performed. A 10 cm long submucosal tunnel is created using TT knife in spray coagulation mode, effect 2, 50 W. Coagulation of big or bleeding vessels is performed using coagrasper in softcoagulation mode effect 5, 80 W.

granular cell tumor is performed using IT knife 2 and TT knife, in spray coagulation mode, effect 2, 50 W and endocut mode effect 2. The granular cell tumor is resected, from the luminal side, with the covering mucosa, using IT knife 2 and TT knife. The 2 mucosal defects are closed with clips.

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Discussion

Endoscopic submucosal tunnel dissection (ESTD) was shown to be feasible, reliable and safe, enabling complete resection of an esophageal granular cell tumor (GCT) in our patient. Granular cell tumors are rare, usually benign and can occur in any part of the body, having the risk of malignant degeneration in only 1–2% of the cases [1]. In the esophagus, GCTs are usually restricted to the submucosa, but in some cases, they can also involve the mucosal or muscularis propria layers [2]. Histologically, GCTs have neurogenic origin, arising from the Schwann cells that are located in the submucosal neuronal plexus of the esophagus [3]. As the risk of malignization is low, a conservative approach is advocated in most cases [4]. These tumors should be removed when symptomatic, when significantly increase in size or when atypical histological or ultrasonographic features are found [4]. If small, GCTs are usually removed through conventional endoscopic mucosal resection, being minimally invasive surgery an option for large lesions [5]. Endoscopic submucosal dissection (ESD) was seldom described for treatment of GCT [2]. The technique of endoscopic submucosal tunnel dissection (ESTD) was recently described for the resection of submucosal gastroesophageal lesions, namely GISTs, leiomyoma and aberrant pancreas [6,7]. As in the case of peroral endoscopic myotomy for the treatment of achalasia, ESTD is based on mucosal incision, creation of a submucosal tunnel and closing of mucosal defects with clips [8]. To our knowledge, ESTD was never reported for the resection of esophageal GCTs. In our patient, poor lifting after submucosal injection precluded conventional ESD and a decision for ESTD was made based on the co-morbilities associated with surgical resection. Flexible endoscopy with biopsies is the mainstay for the diagnosis of esophageal GCT [4]. Like in more than 80% of esophageal GCTs, in our patient the histological diagnosis could be done with conventional biopsy samples [5]. Endoscopic ultrasound was performed to characterize the lesion, to provide additional information on the layer of origin, evaluate the tumor extension and to excluded muscularis propria involvement that could be the cause of the poor submucosal lifting. In ESTD, a submucosal tunnel is performed. The tangential dissection enables an accurated approach to the submucosal fibers avoiding the muscle layer and reducing the risk of perforation. As it was not possible to completely dissect the GCT from the mucosal layer, a resection including the mucosa was performed as described previously for ESTD of aberrant pancreas [9]. En bloc resection was

Please cite this article as: Baldaque-Silva F, et al. Endoscopic Submucosal Tunnel Dissection for Esophageal Granular Cell Tumor Resection. Video Journal and Encyclopedia of GI Endoscopy (2013), http://dx.doi.org/10.1016/j.vjgien.2013.05.004

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accomplished, being the 25 mm long lesion retrieved through the esophageal lumen. The 2 esophageal mucosal orifices were closed with clips as described previously for ESTD of aberrant pancreas [9]. The patient started oral diet 1 day after ESTD and was discharged without any complications, being asymptomatic at the 2 and 6 months follow-up. Histologically, the resected lesion was well-circumscribed, composed of fusiform cells with abundant eosinophilic cytoplasm separated by collagenous septae into distinct clusters. No atypia or mitotic figures were observed, nor there was any evidence of necrosis. Immunohistochemically, there was a strong and diffuse positivity for S-100 protein, confirming the previous diagnosis of GCT. This case highlights the usefulness of ESTD. This technique may be considered in cases of esophageal GCT with indication for resection, when poor submucosal lifting hampers conventional ESD.

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Scripted voiceover

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51 year-old male patient. Erosive esophagitis. Esophageal subepithelial lesion with increased size in the follow-up. EUS shows a lesion restricted to the submucosa. Conventional biopsy specimens are positive for granular cell tumor. Poor submucosal lifting precludes ESD resection. A decision for endoscopic submucosal tunnel dissection is made. This is the endoscopy view, showing the subepithelial lesion in the distal esophagus and signs of reflux esophagitis and scars from previous biopsies. Endoscopic ultrasound is performed using a radial scope. The lesion is located in the submucosal layer, is heterogeneous and is in deep contact with the mucosal and muscularis propria layers. The transverse diameters are 12 and 8 mm. At this level the muscularis propria is 1 mm thick The esophagus is rinsed with saline and chlorhexidine using a catheter spray. A pseudopolyp is created 5 cm above the lesion, using a sclerotherapy needle and a saline solution with diluted adrenalin and methylene blue. Then, a longitudinal incision is made using TT knife in endocut mode effect 2. Using continuous endoscope-knife traction a 5 cm long incision is performed. Injection with saline expands the submucosa. This is important, because, contrary to POEM, in this case the muscularis propria is thin, increasing the risk of perforation. In order to get inside the submucosal space, a dedicated conic and short cap is used. Coagulation of big or bleeding vessels is performed using a coagrasper in softcoagulation mode effect 5, 80 W. A 10 cm long submucosal tunnel is created using TT knife and IT knife 2, in spray coagulation mode, effect 2, 50 W. Gentle movements are important to avoid injury to the mucosa and muscularis propria layers

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Dissection of the lesion submucosal side is performed using IT knife 2 and TT knife, in spray coagulation mode, effect 2, 50 W. It is impossible to dissect completely the lesion from the mucosal layer. That is the reason why we resected the granular cell tumor, from the luminal side with the covering mucosa. This was done using IT knife 2 and TT knife on endocut mode effect 2 The lesion is retrieved using a tripod The 2 mucosal defects are closed with clips We start closing the distal mucosal defect. First, a clip is placed in the distal side of the mucosal defect in order to create a fold. After this, the remaining clips are placed one by one. As it can be seen, the cap is important here to deviate the previous clip, before placing the next one. These clips cannot be opened after closing, so care should be taken in positioning them before full closure. Here a slim gastroscope is used to confirm the closure of the 2 mucosal defects. The retrieved specimen measures 25 mm Histological examination with hematoxylin and eosin, shows a well-circumscribed subepithelial lesion, composed of fusiform cells with abundant eosinophilic cytoplasm separated by collagenous septae into distinct clusters. At 100 times magnification, no atypia, mitotic figures or necrosis are observed Immunohistochemically, there is a strong positivity for S-100. This is S-100 immunohistochemistry, with 40 times magnification The patient remained asymptomatic at the 2 and 6 months follow-up. At the 6 months follow-up, some clips used in the proximal mucosal defect were still in place. Esophageal granular cells tumors should be resected when symptomatic, atypical or increasing in size. In cases of poor submucosal lifting, endoscopic submucosal tunnel dissection enables a safe and accurate resection.

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Conflict of interest Authors declare no conflicts of interest.

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Acknowledgments

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Authors would like to thank Dr. Kenichi Goda, from the Department of Endoscopy, Jikei University School of Medicine, Tokyo, Japan, for providing a dedicated endoscopic cap for this procedure.

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References [1] Ordonez NG. Granular cell tumor: a review and update. Adv Anat Pathol 1999;6(4):186–203. [2] Nakajima M, et al. Esophageal granular cell tumor successfully Q7 resected by endoscopic submucosal dissection. Esophagus 2011;8(3):203–7. [3] Fisher ER, Wechsler H. Granular cell myoblastoma—a misnomer. Electron microscopic and histochemical evidence concerning its Schwann cell derivation and nature (granular cell schwannoma). Cancer 1962;15:936–54.


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[7] Gong W, et al. Preliminary experience of endoscopic submucosal tunnel dissection for upper gastrointestinal submucosal tumors. Endoscopy 2012;44(3):231–5. [8] Inoue H, et al. Peroral endoscopic myotomy (POEM) for esophageal achalasia. Endoscopy 2010;42(4):265–71. [9] Inoue H, et al. Submucosal endoscopic tumor resection for Q8 subepithelial tumors in the esophagus and cardia. Endoscopy 2012;44(3):225–30.

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