- Email: [email protected]
Endoscopic diagnosis and treatment of gastric glomus tumors
Endoscopic diagnosis and treatment of gastric glomus tumors Yiqun Zhang, MD, Pinghong Zhou, MD, Meidong Xu, MD, Weifeng Chen, MD, Quanlin Li, MD, Yuan Ji, MD, Liqing Yao, MD Shanghai, People’s Republic of China
Background: Although gastric glomus tumors are usually benign lesions, occasional malignant transformation has been reported. Thus, complete resection of the gastric glomus tumor is necessary. Objective: To provide a better understanding of the endoscopic features of this rare entity with an emphasis on its diagnosis and treatment. Design: Retrospective case series. Setting: Academic medical center. Patients: Six patients (2 men, 4 women; median age 48 years) received a diagnosis of gastric glomus tumor and were treated. Interventions: Endoscopic diagnosis and resection. Main Outcome Measurements: Endoscopic features, resection success, adverse events, and follow-up endoscopy. Results: Gastric glomus tumors do not exhibit specific features on gastroscopy and EUS that distinguish them from other gastric submucosal tumors. Endoscopic submucosal enucleation was successful in 5 patients. In one patient, the operation had to be discontinued because of significant bleeding during the procedure. The mean tumor size was 19.8 ⫾ 6.2 mm (range 12-30 mm). Perforation occurred in 1 patient and was successfully managed with hemoclips. No local recurrence was observed during follow-up (mean duration 9 ⫾ 5.1 months, range 3-17 months). Limitations: Small number of patients (N ⫽ 6), limited follow-up, retrospective study. Conclusions: Diagnosis of gastric glomus tumors is difficult when based only on features derived from gastroscopy and EUS. Endoscopic submucosal enucleation is a feasible and safe procedure with which to diagnose and treat this lesion. However, further investigation and comparative studies are required to confirm the safety and efficacy of this method.
Glomus tumors are always found in peripheral soft tissues such as the dermis or subungual region,1 but can occur anywhere in the GI tract, especially in the gastric antrum.2 Gastric glomus tumors are rare; their frequency is estimated at approximately 1% that of GI stromal tumors.3 To date, only 130 cases of gastric glomus tumor have been reported in the literature.4 They are most commonly described as solitary, well-defined submucosal lesions of the stomach. It is difficult to distinguish these lesions preoperatively from other gastric submucosal tumors because of the poor characterization of specific clinical or endoscopic features and their intramural location, which precludes
diagnosis by conventional endoscopic tissue biopsy. Although gastric glomus tumors are usually benign lesions, malignant transformation and multiple lesions have occasionally been reported.5,6 Thus, gastric glomus tumors pose an important but tremendous diagnostic and therapeutic challenge for the endoscopists. Conservative local resection by open or laparoscopic surgery is usually sufficient therapy for glomus tumors of the stomach. An alternative strategy would be to avoid invasive surgical procedures by extending the use of endoscopy to the evaluation and treatment of gastric glomus tumors. We hypothesized that by applying the recently
Abbreviations: EFTR, endoscopic full-thickness resection; ESD, endoscopic submucosal dissection.
doi:10.1016/j.gie.2010.10.023
DISCLOSURE: The authors disclosed no financial relationships relevant to this publication. This work was supported by grants from the Key Project of Shanghai Sci-Tech Research & Development Program (09DZ1950102) and the Specific Fund for Health Research in the Public Interst of China (200902002-3). Copyright © 2011 by the American Society for Gastrointestinal Endoscopy 0016-5107/$36.00
www.giejournal.org
Received August 25, 2010. Accepted October 14, 2010. Institute of Endoscopy (Y.Z., P.Z., M.X., W.C., Q.L., L.Y.), Department of Pathology (Y.J.), Zhongshan Hospital, Fudan University, Shanghai, People’s Republic of China. Reprint requests: Liqing Yao, MD, Institute of Endoscopy, Zhongshan Hospital, Fudan University, 180 FengLin Road, Shanghai 200032, People’s Republic of China.
Volume 73, No. 2 : 2011 GASTROINTESTINAL ENDOSCOPY
371
Endoscopic diagnosis and treatment of gastric glomus tumors
developed technique of endoscopic submucosal dissection (ESD), we could develop a method to diagnose and resect gastric glomus tumors endoscopically. The purpose of this report is to provide a better understanding of the endoscopic features of these rare tumors with an emphasis on diagnosis and treatment.
PATIENTS AND METHODS The cases were collected retrospectively by searching the histologic diagnosis database of gastroscopy and gastrectomy specimens at Zhongshan Hospital between January 2009 and July 2010. Six patients (2 men, 4 women; mean age 48.7 years) with gastric glomus tumors received a diagnosis and were treated at our institute. All patients reported epigastric discomfort, and lesions were found on endoscopy. In all 6 patients, EUS was performed before treatment with a UM-2R system (Olympus Optical, Tokyo, Japan) to evaluate the origin and size of the tumors. Informed consent for all procedures, including endoscopic resection, was obtained from each patient. We obtained institutional review board approval for the study. To dissect the tumor, endoscopic submucosal enucleation was attempted with a single-channel gastroscope (GIF-H260; Olympus Optical) and an insulated-tip electrosurgical knife (KD-611L; Olympus Optical) or hook-knife (KD-620LR; Olympus Optical). We used the ERBE ICC-200 high-frequency generator (ERBE Elektromedizin, Tübingen, Germany). A transparent cap (D-201-11304; Olympus Optical) was attached to the tip of the gastroscope to provide direct views of the submucosal layer. Patients were treated while under general anesthesia. Marking dots were placed on the normal mucosa approximately 5 mm from the tumor margin. After submucosal injection of saline solution with a small amount of indigo carmine and epinephrine (0.0005%), the mucosa was incised outside the marking dots. Direct dissection of the submucosal layer beneath the tumor was then performed under direct vision to achieve complete en bloc resection of the specimen. The tumor was dissected along the capsule, and saline solution was injected repeatedly during the dissection when necessary. The resultant artificial ulcer was managed routinely with argon plasma coagulation to prevent delayed bleeding, and hemoclips were used to close the deeply dissected areas as needed.
Pathologic evaluation Complete resection was defined as a resected specimen in which both lateral and vertical margins were tumor free. Incomplete resection was defined as a specimen with histologically positive margins.
RESULTS Glomus tumors of the stomach, as viewed by gastroscopy in our study, appeared as well circumscribed sub372 GASTROINTESTINAL ENDOSCOPY
Volume 73, No. 2 : 2011
Zhang et al
mucosal masses with normal overlying mucosa (Fig. 1A). The EUS features of the lesion included a sharply demarcated mass in the fourth layer of the gastric wall and a heterogeneous hypoechoic pattern with internal hyperechoic spots and a few tubular structures (Fig. 1B). Patient characteristics, lesion features, and clinical outcomes are summarized in Table 1. Mean patient age was 48.7 ⫾ 10.6 years (range 32–58 years), and mean tumor size was 19.8 ⫾ 6.2 mm (range 12–30 mm). The tumors in 5 patients were resected completely (Fig. 1C, D), whereas 1 patient had a tumor remnant. Histopathologically, glomus tumors were well-circumscribed lesions consisting of tight convolutes of capillary-sized vessels surrounded by collars of glomus cells set in hyalinized or myxoid stroma. Some areas exhibited an organoid or epithelioid pattern of growth. The tumor cells showed rounded nuclei set off from amphophilic or eosinophilic cytoplasm (Fig. 1E). Endoscopic submucosal enucleation was successful in 5 patients. In 1 patient, significant bleeding from the surrounding tissue, which was rich in microvessels, blurred the endoscopic view, making en bloc resection difficult. Control of bleeding with argon plasma coagulation and hemoclips was unsuccessful; therefore, piecemeal resection was performed. The incompletely resected sample was sent to the Department of Pathology. One week later, a gastric glomus tumor was confirmed; in addition, tumor cells, which showed mild atypia, were found in the deep margin. This patient underwent subtotal gastrectomy 10 days after endoscopic submucosal enucleation. In the resected gastric sample, a superficial ulcer was detected in the antrum, and small nodules of residual tumor cells were found in the muscular layer. Tumor cells showed mild atypia with no mitotic figures observed on high power. Perforation occurred in another patient because the tumor adhered tightly to the serosa. The tumor and serosa were resected simultaneously to achieve complete resection, and the perforation was successfully managed by hemoclips. The duration of follow-up ranged from 3 to 17 months (mean duration 9 ⫾ 5.1 months). All the patients underwent gastroscopic examination (Fig. 1F). No local recurrence was observed during follow-up.
DISCUSSION Glomus tumors are rare and benign lesions in the stomach, first reported by De Bussacher in 1948.7 On endoscopy, the tumor appears as a submucosal lesion with a smooth surface, although erosion and ulceration of the overlying mucosa are common. Most gastric glomus tumors occur in the antrum portion of the stomach.8 In this study, 66.7% lesions were located in the antrum. When a gastric glomus tumor is found at gastroscopy, the barrier to clinical management lies in the uncertainty about the histopathologic nature of the tumor. Most gastric glomus tumors are benign, but malignant tumors, which can metastasize to the liver or other organs/tissues, have www.giejournal.org
Zhang et al
Endoscopic diagnosis and treatment of gastric glomus tumors
Figure 1. Endoscopic submucosal enucleation of a glomus tumor located in the antrum in a 49-year-old woman (patient 3). A, Glomus tumor (22-mm diameter). B, EUS shows a mass in the fourth layer of the gastric wall. C, View of the submucosal layer and direct dissection of the tumor with the use of a soft transparent hood. D, Completely resected specimen. E, Microscopic examination of the completely resected specimen reveals tight convolutes of capillary-sized vessels surrounded by collars of glomus cells set in hyalinized or myxoid stroma with negative margins (H&E, orig. mag. ⫻40). F, Endoscopic findings of scar 2 months after endoscopic submucosal enucleation.
www.giejournal.org
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Endoscopic diagnosis and treatment of gastric glomus tumors
Zhang et al
TABLE 1. Patient characteristics and outcomes Patient
Age(y)
Sex
Tumor size (mm)
Gastric site
Complications
Resection
1
32
F
12
Antrum
—
Complete
2
40
M
20
Body
Perforation
Complete
3
49
F
22
Antrum
—
Complete
4
58
F
15
Body
—
Complete
5
56
F
20
Fundus
—
Complete
6
57
M
30
Antrum
Bleeding
Incomplete
Complete, lateral and vertical margins free of tumor.
been reported.9 Gastric glomus tumors do not exhibit specific features on EUS that distinguish them from other submucosal tumors.10 Studies have reported diagnosis of these tumors by power Doppler endosonography and EUS-guided FNA11-14; however, these methods cannot provide adequate histopathologic evaluation. Complete resection of lesion is necessary for its accurate characterization and patient treatment. In the past, open or laparoscopic surgery was the main treatment option for this kind of lesion. However, surgery is highly invasive and can lead to a decrease in the patient’s quality of life compared with simple endoscopic resection. Endoscopic resection can prevent scars caused by abdominal incisions, reduce the inflammatory response and perceived pain associated with surgical trauma, and lower the risk of postoperative infection. The gastric wall is partially resected during laparoscopic surgery, which changes the anatomic structure of the stomach. Esophagogastric anastomosis is required when the tumor is near the gastric cardia, which usually provokes reflux. However, endoscopic resection can be performed to preserve gastric function as much as possible.15,16 Because of the success of ESD for en bloc resection of early gastric cancer,17 studies have begun to find the benefits of endoscopic resection for submucosal tumors.18,19 At our institute, endoscopic submucosal enucleation in this study was performed to remove many gastric submucosal tumors. Postoperative histopathologic diagnoses of the recovered specimens included leiomyomas, stromal tumors, and glomus tumors. In this study, we demonstrated that endoscopic submucosal enucleation is a safe, effective, and minimally invasive procedure for treating gastric glomus tumors. Endoscopic submucosal enucleation was successful in 5 of 6 patients (83.3%); however, the operation had to be discontinued in 1 patient because of significant bleeding during the procedure. In this case, the tumor appears as a poorly circumscribed lesion. Microscopically, proliferation of glomus cells in vessels at the periphery of the tumor may have contributed to the bleeding. Among the remaining 5 patients, complete resection was successful without 374 GASTROINTESTINAL ENDOSCOPY
Volume 73, No. 2 : 2011
serious complication, except in 1 patient in whom a small perforation occurred because of tight adhesion of the tumor to the serosa. Although we thought perforation might be unavoidable, endoscopic submucosal enucleation was attempted. The perforation was closed immediately with hemoclips, and this procedure was called an endoscopic full-thickness resection (EFTR). After careful monitoring for 5 days, the patient recovered and was discharged. EFTR was attempted based on the development of the ESD technique and EFTR suturing devices. Our study challenges the prevailing idea that gastric perforation is a serious complication. During EFTR, deliberate gastric perforation is performed for complete resection of the tumor. New tools and devices for endoscopic closure of these perforations have been developed.20-22 EFTR has been shown to be an efficacious, safe, and minimally invasive procedure for patients with gastric submucosal tumors originating from the muscularis propria layer.23,24 The development of EFTR may extend the indications for endoscopic resection. Follow-up ranged from 3 to 17 months (mean duration 9 ⫾ 5.1 months), during which local recurrence was not observed in any patient by gastroscopy. However, because of the limited number of patients and relatively short follow-up duration, we realize that much longer follow-up may be needed to evaluate long-term results of endoscopic submucosal enucleation of gastric glomus tumors. In conclusion, we showed that gastric glomus tumors are difficult to diagnose when relying only on features derived from gastroscopy and EUS. Endoscopic submucosal enucleation seems to be a feasible and safe procedure with which to diagnose and treat these lesions. However, controlled clinical trials involving more patients and longer follow-up are needed to confirm our results. REFERENCES 1. Tsuneyoshi M, Enjoji M. Glomus tumor: a clinicopathologic and electron microscopic study. Cancer 1982;50:1601-7. 2. Chou HP, Tiu CM, Chen JD, et al. Glomus tumor in the stomach. Abdom Imaging 2010;35:390-2.
www.giejournal.org
Zhang et al 3. Miettinen M, Paal E, Lasota J, et al. Gastrointestinal glomus tumors: a clinicopathologic, immunohistochemical, and molecular genetic study of 32 cases. Am J Surg Pathol 2002;26:301-11. 4. Lee HW, Lee JJ, Yang DH, et al. A clinicopathologic study of glomus tumor of the stomach. J Clin Gastroenterol 2006;40:717-20. 5. Folpe AL, Fanburg-Smith JC, Miettinen M, et al. Atypical and malignant glomus tumors: analysis of 52 cases, with a proposal for the reclassification of glomus tumors. Am J Surg Pathol 2001;25:1-12. 6. Haque S, Modlin IM, West AB. Multiple glomus tumors of the stomach with intravascular spread. Am J Surg Pathol 1992;16:291. 7. De Busscher G. Les anatomoses arterioveineuses de l’estomac [French]. Acta Neurol Morphol 1948;6:87-105. 8. Agawa H, Matsushita M, Nishio A, et al. Gastric glomus tumor. Gastrointest Endosc 2002;56:903. 9. Bray AP, Wong NA, Narayan S. Cutaneous metastasis from gastric glomus tumour. Clin Exp Dermatol 2009;34:e719-21. 10. Shin HM, Ryu DY, Lee D, et al. A case of glomus tumor of the stomach: role of endoscopic ultrasonography. Korean J Gastrointest Endosc 2000; 21:855-8. 11. Yan SL, Yeh YH, Chen CH, et al. Gastric glomus tumor: a hypervascular submucosal tumor on power Doppler endosonography. J Clin Ultrasound 2007;35:164-8. 12. Vinette-Leduc D, Yazdi HM. Fine-needle aspiration biopsy of a glomus tumor of the stomach. Diagn Cytopathol 2001;24:340-2. 13. Debol SM, Stanley MW, Mallery S, et al. Glomus tumor of the stomach: cytologic diagnosis by endoscopic ultrasound-guided fine-needle aspiration. Diagn Cytopathol 2003;28:316-21. 14. Gu M, Nguyen PT, Cao S, et al. Diagnosis of gastric glomus tumor by endoscopic ultrasound-guided fine needle aspiration biopsy. A case report with cytologic, histologic and immunohistochemical studies. Acta Cytol 2002;46:560-6.
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Endoscopic diagnosis and treatment of gastric glomus tumors 15. Marescaux J, Dallemagne B, Perretta S, et al. Surgery without scars: report of transluminal cholecystectomy in a human being. Arch Surg 2007;142:823-6. 16. Wang L, Ren W, Fan CQ, et al. Full-thickness endoscopic resection of nonintracavitary gastric stromal tumors: a novel approach. Surg Endosc. Epub 2010 Jun 30. 17. Gotoda T. A large endoscopic resection by endoscopic submucosal dissection procedure for early gastric cancer. Clin Gastroenterol Hepatol 2005;3:S71-3. 18. Park YS, Park SW, Kim TI, et al. Endoscopic enucleation of upper-GI submucosal tumors by using an insulated-tip electrosurgical knife. Gastrointest Endosc 2004;59:409-15. 19. Hoteya S, Iizuka T, Kikuchi D, et al. Endoscopic submucosal dissection for gastric submucosal tumor, endoscopic sub-tumoral dissection. Dig Endosc 2009;21:266-9. 20. Kaehler G, Grobholz R, Langner C, et al. A new technique of endoscopic full-thickness resection using a flexible stapler. Endoscopy 2006;38: 86-9. 21. Kantsevoy SV. Endoscopic full-thickness resection: new minimally invasive therapeutic alternative for GI-tract lesions. Gastrointest Endosc 2006;64:90-1. 22. von Renteln D, Schmidt A, Riecken B, et al. Gastric full-thickness suturing during EMR and for treatment of gastric-wall defects (with video). Gastrointest Endosc 2008,67:738-44. 23. Zhou PH, Yao LQ, Qin XY, et al. Endoscopic full-thickness resection without laparoscopic assistance for gastric submucosal tumors originated from muscularis propria layer. Chin J Dig Endosc 2009;26:617-20. 24. von Renteln D, Riecken B, Walz B, et al. Endoscopic GIST resection using FlushKnife ESD and subsequent perforation closure by means of endoscopic full-thickness suturing. Endoscopy 2008,40(Suppl 2): E224-5.
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Background: Although gastric glomus tumors are usually benign lesions, occasional malignant transformation has been reported. Thus, complete resection of the gastric glomus tumor is necessary. Objective: To provide a better understanding of the endoscopic features of this rare entity with an emphasis on its diagnosis and treatment. Design: Retrospective case series. Setting: Academic medical center. Patients: Six patients (2 men, 4 women; median age 48 years) received a diagnosis of gastric glomus tumor and were treated. Interventions: Endoscopic diagnosis and resection. Main Outcome Measurements: Endoscopic features, resection success, adverse events, and follow-up endoscopy. Results: Gastric glomus tumors do not exhibit specific features on gastroscopy and EUS that distinguish them from other gastric submucosal tumors. Endoscopic submucosal enucleation was successful in 5 patients. In one patient, the operation had to be discontinued because of significant bleeding during the procedure. The mean tumor size was 19.8 ⫾ 6.2 mm (range 12-30 mm). Perforation occurred in 1 patient and was successfully managed with hemoclips. No local recurrence was observed during follow-up (mean duration 9 ⫾ 5.1 months, range 3-17 months). Limitations: Small number of patients (N ⫽ 6), limited follow-up, retrospective study. Conclusions: Diagnosis of gastric glomus tumors is difficult when based only on features derived from gastroscopy and EUS. Endoscopic submucosal enucleation is a feasible and safe procedure with which to diagnose and treat this lesion. However, further investigation and comparative studies are required to confirm the safety and efficacy of this method.
Glomus tumors are always found in peripheral soft tissues such as the dermis or subungual region,1 but can occur anywhere in the GI tract, especially in the gastric antrum.2 Gastric glomus tumors are rare; their frequency is estimated at approximately 1% that of GI stromal tumors.3 To date, only 130 cases of gastric glomus tumor have been reported in the literature.4 They are most commonly described as solitary, well-defined submucosal lesions of the stomach. It is difficult to distinguish these lesions preoperatively from other gastric submucosal tumors because of the poor characterization of specific clinical or endoscopic features and their intramural location, which precludes
diagnosis by conventional endoscopic tissue biopsy. Although gastric glomus tumors are usually benign lesions, malignant transformation and multiple lesions have occasionally been reported.5,6 Thus, gastric glomus tumors pose an important but tremendous diagnostic and therapeutic challenge for the endoscopists. Conservative local resection by open or laparoscopic surgery is usually sufficient therapy for glomus tumors of the stomach. An alternative strategy would be to avoid invasive surgical procedures by extending the use of endoscopy to the evaluation and treatment of gastric glomus tumors. We hypothesized that by applying the recently
Abbreviations: EFTR, endoscopic full-thickness resection; ESD, endoscopic submucosal dissection.
doi:10.1016/j.gie.2010.10.023
DISCLOSURE: The authors disclosed no financial relationships relevant to this publication. This work was supported by grants from the Key Project of Shanghai Sci-Tech Research & Development Program (09DZ1950102) and the Specific Fund for Health Research in the Public Interst of China (200902002-3). Copyright © 2011 by the American Society for Gastrointestinal Endoscopy 0016-5107/$36.00
www.giejournal.org
Received August 25, 2010. Accepted October 14, 2010. Institute of Endoscopy (Y.Z., P.Z., M.X., W.C., Q.L., L.Y.), Department of Pathology (Y.J.), Zhongshan Hospital, Fudan University, Shanghai, People’s Republic of China. Reprint requests: Liqing Yao, MD, Institute of Endoscopy, Zhongshan Hospital, Fudan University, 180 FengLin Road, Shanghai 200032, People’s Republic of China.
Volume 73, No. 2 : 2011 GASTROINTESTINAL ENDOSCOPY
371
Endoscopic diagnosis and treatment of gastric glomus tumors
developed technique of endoscopic submucosal dissection (ESD), we could develop a method to diagnose and resect gastric glomus tumors endoscopically. The purpose of this report is to provide a better understanding of the endoscopic features of these rare tumors with an emphasis on diagnosis and treatment.
PATIENTS AND METHODS The cases were collected retrospectively by searching the histologic diagnosis database of gastroscopy and gastrectomy specimens at Zhongshan Hospital between January 2009 and July 2010. Six patients (2 men, 4 women; mean age 48.7 years) with gastric glomus tumors received a diagnosis and were treated at our institute. All patients reported epigastric discomfort, and lesions were found on endoscopy. In all 6 patients, EUS was performed before treatment with a UM-2R system (Olympus Optical, Tokyo, Japan) to evaluate the origin and size of the tumors. Informed consent for all procedures, including endoscopic resection, was obtained from each patient. We obtained institutional review board approval for the study. To dissect the tumor, endoscopic submucosal enucleation was attempted with a single-channel gastroscope (GIF-H260; Olympus Optical) and an insulated-tip electrosurgical knife (KD-611L; Olympus Optical) or hook-knife (KD-620LR; Olympus Optical). We used the ERBE ICC-200 high-frequency generator (ERBE Elektromedizin, Tübingen, Germany). A transparent cap (D-201-11304; Olympus Optical) was attached to the tip of the gastroscope to provide direct views of the submucosal layer. Patients were treated while under general anesthesia. Marking dots were placed on the normal mucosa approximately 5 mm from the tumor margin. After submucosal injection of saline solution with a small amount of indigo carmine and epinephrine (0.0005%), the mucosa was incised outside the marking dots. Direct dissection of the submucosal layer beneath the tumor was then performed under direct vision to achieve complete en bloc resection of the specimen. The tumor was dissected along the capsule, and saline solution was injected repeatedly during the dissection when necessary. The resultant artificial ulcer was managed routinely with argon plasma coagulation to prevent delayed bleeding, and hemoclips were used to close the deeply dissected areas as needed.
Pathologic evaluation Complete resection was defined as a resected specimen in which both lateral and vertical margins were tumor free. Incomplete resection was defined as a specimen with histologically positive margins.
RESULTS Glomus tumors of the stomach, as viewed by gastroscopy in our study, appeared as well circumscribed sub372 GASTROINTESTINAL ENDOSCOPY
Volume 73, No. 2 : 2011
Zhang et al
mucosal masses with normal overlying mucosa (Fig. 1A). The EUS features of the lesion included a sharply demarcated mass in the fourth layer of the gastric wall and a heterogeneous hypoechoic pattern with internal hyperechoic spots and a few tubular structures (Fig. 1B). Patient characteristics, lesion features, and clinical outcomes are summarized in Table 1. Mean patient age was 48.7 ⫾ 10.6 years (range 32–58 years), and mean tumor size was 19.8 ⫾ 6.2 mm (range 12–30 mm). The tumors in 5 patients were resected completely (Fig. 1C, D), whereas 1 patient had a tumor remnant. Histopathologically, glomus tumors were well-circumscribed lesions consisting of tight convolutes of capillary-sized vessels surrounded by collars of glomus cells set in hyalinized or myxoid stroma. Some areas exhibited an organoid or epithelioid pattern of growth. The tumor cells showed rounded nuclei set off from amphophilic or eosinophilic cytoplasm (Fig. 1E). Endoscopic submucosal enucleation was successful in 5 patients. In 1 patient, significant bleeding from the surrounding tissue, which was rich in microvessels, blurred the endoscopic view, making en bloc resection difficult. Control of bleeding with argon plasma coagulation and hemoclips was unsuccessful; therefore, piecemeal resection was performed. The incompletely resected sample was sent to the Department of Pathology. One week later, a gastric glomus tumor was confirmed; in addition, tumor cells, which showed mild atypia, were found in the deep margin. This patient underwent subtotal gastrectomy 10 days after endoscopic submucosal enucleation. In the resected gastric sample, a superficial ulcer was detected in the antrum, and small nodules of residual tumor cells were found in the muscular layer. Tumor cells showed mild atypia with no mitotic figures observed on high power. Perforation occurred in another patient because the tumor adhered tightly to the serosa. The tumor and serosa were resected simultaneously to achieve complete resection, and the perforation was successfully managed by hemoclips. The duration of follow-up ranged from 3 to 17 months (mean duration 9 ⫾ 5.1 months). All the patients underwent gastroscopic examination (Fig. 1F). No local recurrence was observed during follow-up.
DISCUSSION Glomus tumors are rare and benign lesions in the stomach, first reported by De Bussacher in 1948.7 On endoscopy, the tumor appears as a submucosal lesion with a smooth surface, although erosion and ulceration of the overlying mucosa are common. Most gastric glomus tumors occur in the antrum portion of the stomach.8 In this study, 66.7% lesions were located in the antrum. When a gastric glomus tumor is found at gastroscopy, the barrier to clinical management lies in the uncertainty about the histopathologic nature of the tumor. Most gastric glomus tumors are benign, but malignant tumors, which can metastasize to the liver or other organs/tissues, have www.giejournal.org
Zhang et al
Endoscopic diagnosis and treatment of gastric glomus tumors
Figure 1. Endoscopic submucosal enucleation of a glomus tumor located in the antrum in a 49-year-old woman (patient 3). A, Glomus tumor (22-mm diameter). B, EUS shows a mass in the fourth layer of the gastric wall. C, View of the submucosal layer and direct dissection of the tumor with the use of a soft transparent hood. D, Completely resected specimen. E, Microscopic examination of the completely resected specimen reveals tight convolutes of capillary-sized vessels surrounded by collars of glomus cells set in hyalinized or myxoid stroma with negative margins (H&E, orig. mag. ⫻40). F, Endoscopic findings of scar 2 months after endoscopic submucosal enucleation.
www.giejournal.org
Volume 73, No. 2 : 2011 GASTROINTESTINAL ENDOSCOPY
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Endoscopic diagnosis and treatment of gastric glomus tumors
Zhang et al
TABLE 1. Patient characteristics and outcomes Patient
Age(y)
Sex
Tumor size (mm)
Gastric site
Complications
Resection
1
32
F
12
Antrum
—
Complete
2
40
M
20
Body
Perforation
Complete
3
49
F
22
Antrum
—
Complete
4
58
F
15
Body
—
Complete
5
56
F
20
Fundus
—
Complete
6
57
M
30
Antrum
Bleeding
Incomplete
Complete, lateral and vertical margins free of tumor.
been reported.9 Gastric glomus tumors do not exhibit specific features on EUS that distinguish them from other submucosal tumors.10 Studies have reported diagnosis of these tumors by power Doppler endosonography and EUS-guided FNA11-14; however, these methods cannot provide adequate histopathologic evaluation. Complete resection of lesion is necessary for its accurate characterization and patient treatment. In the past, open or laparoscopic surgery was the main treatment option for this kind of lesion. However, surgery is highly invasive and can lead to a decrease in the patient’s quality of life compared with simple endoscopic resection. Endoscopic resection can prevent scars caused by abdominal incisions, reduce the inflammatory response and perceived pain associated with surgical trauma, and lower the risk of postoperative infection. The gastric wall is partially resected during laparoscopic surgery, which changes the anatomic structure of the stomach. Esophagogastric anastomosis is required when the tumor is near the gastric cardia, which usually provokes reflux. However, endoscopic resection can be performed to preserve gastric function as much as possible.15,16 Because of the success of ESD for en bloc resection of early gastric cancer,17 studies have begun to find the benefits of endoscopic resection for submucosal tumors.18,19 At our institute, endoscopic submucosal enucleation in this study was performed to remove many gastric submucosal tumors. Postoperative histopathologic diagnoses of the recovered specimens included leiomyomas, stromal tumors, and glomus tumors. In this study, we demonstrated that endoscopic submucosal enucleation is a safe, effective, and minimally invasive procedure for treating gastric glomus tumors. Endoscopic submucosal enucleation was successful in 5 of 6 patients (83.3%); however, the operation had to be discontinued in 1 patient because of significant bleeding during the procedure. In this case, the tumor appears as a poorly circumscribed lesion. Microscopically, proliferation of glomus cells in vessels at the periphery of the tumor may have contributed to the bleeding. Among the remaining 5 patients, complete resection was successful without 374 GASTROINTESTINAL ENDOSCOPY
Volume 73, No. 2 : 2011
serious complication, except in 1 patient in whom a small perforation occurred because of tight adhesion of the tumor to the serosa. Although we thought perforation might be unavoidable, endoscopic submucosal enucleation was attempted. The perforation was closed immediately with hemoclips, and this procedure was called an endoscopic full-thickness resection (EFTR). After careful monitoring for 5 days, the patient recovered and was discharged. EFTR was attempted based on the development of the ESD technique and EFTR suturing devices. Our study challenges the prevailing idea that gastric perforation is a serious complication. During EFTR, deliberate gastric perforation is performed for complete resection of the tumor. New tools and devices for endoscopic closure of these perforations have been developed.20-22 EFTR has been shown to be an efficacious, safe, and minimally invasive procedure for patients with gastric submucosal tumors originating from the muscularis propria layer.23,24 The development of EFTR may extend the indications for endoscopic resection. Follow-up ranged from 3 to 17 months (mean duration 9 ⫾ 5.1 months), during which local recurrence was not observed in any patient by gastroscopy. However, because of the limited number of patients and relatively short follow-up duration, we realize that much longer follow-up may be needed to evaluate long-term results of endoscopic submucosal enucleation of gastric glomus tumors. In conclusion, we showed that gastric glomus tumors are difficult to diagnose when relying only on features derived from gastroscopy and EUS. Endoscopic submucosal enucleation seems to be a feasible and safe procedure with which to diagnose and treat these lesions. However, controlled clinical trials involving more patients and longer follow-up are needed to confirm our results. REFERENCES 1. Tsuneyoshi M, Enjoji M. Glomus tumor: a clinicopathologic and electron microscopic study. Cancer 1982;50:1601-7. 2. Chou HP, Tiu CM, Chen JD, et al. Glomus tumor in the stomach. Abdom Imaging 2010;35:390-2.
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Endoscopic diagnosis and treatment of gastric glomus tumors 15. Marescaux J, Dallemagne B, Perretta S, et al. Surgery without scars: report of transluminal cholecystectomy in a human being. Arch Surg 2007;142:823-6. 16. Wang L, Ren W, Fan CQ, et al. Full-thickness endoscopic resection of nonintracavitary gastric stromal tumors: a novel approach. Surg Endosc. Epub 2010 Jun 30. 17. Gotoda T. A large endoscopic resection by endoscopic submucosal dissection procedure for early gastric cancer. Clin Gastroenterol Hepatol 2005;3:S71-3. 18. Park YS, Park SW, Kim TI, et al. Endoscopic enucleation of upper-GI submucosal tumors by using an insulated-tip electrosurgical knife. Gastrointest Endosc 2004;59:409-15. 19. Hoteya S, Iizuka T, Kikuchi D, et al. Endoscopic submucosal dissection for gastric submucosal tumor, endoscopic sub-tumoral dissection. Dig Endosc 2009;21:266-9. 20. Kaehler G, Grobholz R, Langner C, et al. A new technique of endoscopic full-thickness resection using a flexible stapler. Endoscopy 2006;38: 86-9. 21. Kantsevoy SV. Endoscopic full-thickness resection: new minimally invasive therapeutic alternative for GI-tract lesions. Gastrointest Endosc 2006;64:90-1. 22. von Renteln D, Schmidt A, Riecken B, et al. Gastric full-thickness suturing during EMR and for treatment of gastric-wall defects (with video). Gastrointest Endosc 2008,67:738-44. 23. Zhou PH, Yao LQ, Qin XY, et al. Endoscopic full-thickness resection without laparoscopic assistance for gastric submucosal tumors originated from muscularis propria layer. Chin J Dig Endosc 2009;26:617-20. 24. von Renteln D, Riecken B, Walz B, et al. Endoscopic GIST resection using FlushKnife ESD and subsequent perforation closure by means of endoscopic full-thickness suturing. Endoscopy 2008,40(Suppl 2): E224-5.
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