Endoscopic removal of granular cell tumors

Endoscopic removal of granular cell tumors

in the receptacle prevented the laceration and injury of the stomach and duodenum. The above method, to the best of our knowledge, has not been descri...

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in the receptacle prevented the laceration and injury of the stomach and duodenum. The above method, to the best of our knowledge, has not been described. Whether or not this technique is appropriate for the removal of other sharp foreign bodies remains to be seen. Potential complications during retrieval include loss of the latex receptacle and laceration of the latex and overlying mucosa, and the emergent need for laparotomy must always be anticipated. However, when an overtube is unavailable, this procedure can provide a successful alternative to laparotomy in the stable patient who has suffered sharp foreign body ingestion. REFERENCES 1. Webb WA. Management of foreign bodies of the upper gastrointestinal tract. Gastroenterology 1988;94:204-16. 2. Selivanov V, Sheldon GF, Cello JP, Crass RA. Management of foreign body ingestion. Ann Surg 1984;199:187-91.

CASE REPORTS

Endoscopic removal of granular cell tumors Ichiro Yasuda, Eiichi Tomita, Kazuo Nagura, Youichi Nishigaki, Osamu Yamada, Hideki Kachi,

Case 1 MD MD MD MD MD MD

Granular cell t u m o r s of the gastrointestinal t r a c t are u n c o m m o n . T h e esophagus is the gastrointestinal site most frequently affected; such t u m o r s are rarely seen in the stomach, colon, or rectum. T h e first case of an esophageal t u m o r was reported by Abrikossoff 1 in 1931. Since t h e n a b o u t 200 cases have been described in the literature. T h e lesions are benign in most cases; Strong et al, 2 reported t h a t of 95 cases, only three were malignant. Because until now preoperative diagnosis has been difficult, the s t a n d a r d t r e a t m e n t has been surgical excision. However, this m a y be avoided if diagnosis is made by endoscopic biopsy or other m e t h ods. We report two cases of granular cell t u m o r t h a t were completely removed endoscopically after localization by means of ultrasonography.

From the Departments of Gastroenterology and Clinical Pathology, Gifu Municipal Hospital, Gifu, 500 Japan. Reprint requests: Ichiro Yasuda, MD, Gastroenterology, Gifu Municipal Hospital,7-1 Kashima-cho, Gi[u, 500 Japan 0016-5107/95/4102-016353.00 + 0 GASTROINTESTINAL ENDOSCOPY Copyright | 1995 by the American Society for Gastrointestinal Endoscopy 37/4/55866 VOLUME 41, NO. 2, 1995

3. Barros JL, Caballero A, Rueda JC, Monturiol JM. Foreign body ingestion: management of 167 Cases. World J Surg 1991;15: 783-8. 4. Weisenberger JE. Hazards of eating razor blades. JAMA 1969; 207:1719. 5. Devanesan J, Pisani A, Sherma P, Kazarian KK, Mersheimer WC. Metallic foreign bodies in the stomach. Arch Surg 1977; 112:664-5. 6. Webb WA, McDaniel L, Jones MT. Foreign bodies of the upper GI tract: current management. South Med J 1984;77:1083. 7. Tedesco FJ. Endoscopic removal of foreign bodies using fiberoptic instruments. South Med J 1984;77:1083. 8. DavidoffE, Towne JB. Ingested foreign bodies. N Y State J Med 1975;75:1003. 9. Himad GM, Fischer GJ. Magnetic removal of foreign bodies from the upper gastrointestinal tract. Radiology1977;123:226-7. 10. McCanse DE, Kurchin A, Hinshaw JR. Gastrointestinal foreign bodies. Am J Surg 1981;142:335. 11. MacManus JE. Perforation of the intestine. Am J Surg 1941;53: 393. 12. Witzel L, Scheurer U, Muhlemann A, Halter F. Removal of razor blades from stomach with fiberoptic endoscope. BMJ 1974; 2:539.

A 52-year-old woman presented to our hospital with anorexia. An upper gastrointestinal x-ray series revealed a protruding lesion with bridging folds on the posterior wall of the middle gastric body. Endoscopic examination further revealed the presence of a submucosal tumor with a yellowish surface (Fig. 1). Endoscopic ultrasonography demonstrated a homogeneous, hypoechoic, clearly demarcated 8-ram mass, which was confined to the submucosal layer and separate from the muscularis propria (Fig. 2). A diagnosis of carcinoid tumor, granular cell tumor, or myogenic tumor originating from the muscularis mucosa was suspected but could not be confirmed by the technique used.

Case 2 A 55-year-old man underwent endoscopic examination during a routine checkup. A submucosal tumor with a yellowish surface was revealed in the middle esophagus (Fig. 3). Granular cell tumor was suspected from the biopsy specimens, but the diagnosis was not confirmed. Endoscopic ultrasonography demonstrated a homogeneous, 5-mm, hypoechoic mass in the submucosal layer, separate from the muscularis propria (Fig. 4). In both cases it was difficult to confirm the diagnosis. Because the tumors were separate from the muscularis propria and relatively small in size, it was thought that complete endoscopic removal could be performed safely without leaving residual tissue. Endoscopic excision was performed using a gastrointestinal fiberscope with two channels. After injection of 5 ml of 10% sodium chloride solution containing 0.2 mg of epinephrine (hypertonic saline-epinephrine or HSE solution) into the submucosa around the lesion, the tumor was lifted with grasp forceps inserted through one channel and cut electrically with a high-frequency snare inserted through the other GASTROINTESTINAL ENDOSCOPY 163

Figure 1. Endoscopic photograph demonstrating a yellowish submucosal tumor in the posterior wall of the middle gastric body (case 1).

Figure 3. Endoscopic photograph demonstrating a yellowish submucosal tumor in the middle esophagus (case 2).

Figure 4. Endoscopic ultrasonography demonstrating a Figure 2. Endoscopic ultrasonography demonstrating a

homogeneous, hypoechoic mass in the submucosal layer (case 2).

homogeneous, hypoechoic, clearly demarcated mass in the submucosal layer (case 1).

channel. HSE solution was injected to separate the tumor from the muscularis propria and prevent bleeding. No complication, such as bleeding or perforation, occurred during or after the procedure. Excised specimens showed complete removal of the lesion. Cross-sections showed the tumors to be well-defined, homogeneous, solid, and of yellowish color (Fig. 5). The tumor was 8 mm in diameter in case 1 and 5 mm in case 2, consistent with our ultrasonographic findings. Histologic appearance was similar in both cases: large, polygonal, histiocyte-like cells with coarsely granular, amphophilic cytoplasm (Fig. 6). The granules were positive to periodic acid-Schiff stain, were unaffected by diastase digestion, and also were immunoreactive to NSE and S-100. The diagnosis of granular cell tumor was made. The nuclei were round and uniform, and no mitotic figures were seen. Electron microscopy also performed in case 1 showed the tumor cells to contain abundant, large, autophagic granules surrounded by the basal lamina (Fig. 7A). Higher magnifi164 G A S T R O I N T E S T I N A L E N D O S C O P Y

cation revealed the large granules to consist of membranebound, presumably autophagic vacuoles containing cellular debris, including mitochondria, myeline figures, and vacuoles (Fig. 7B).

DISCUSSION Granular cell t u m o r s (GCTs), which occur infrequently, are probably of Schwann cell origin. 3 T h e y can occur almost anywhere in the b o d y b u t usually affect the tongue, oral cavity, skin, or breasts and are rarely found in the gastrointestinal tract. In fact, only a b o u t 1% of G C T s arise in the gastrointestinal tract. 4 T h e esophagus is the most f r e q u e n t gastrointestinal site, followed by the stomach and the colon. T o our knowledge, only 24 cases of gastric G C T have been rep o r t e d previously. 5-21 Most G C T s of the gastrointestinal t r a c t are submucosal and thus are covered by normal mucosa, with only five previously r e p o r t e d lesions showing mucosal ulceration. & 12, 17, 20, 21 One of these VOLUME 41, NO. 2, 1995

Figure 5. Cross-sections of the resected specimens revealing well-defined, homogeneous, solid, tumor in the submucosa. A, Case 1. B, Case 2.

five patients presented with massive upper gastrointestinal bleeding, 17 and another had concomitant early gastric carcinoma. 21 Granular cell tumors are generally not large, being mostly between 1 and 2 cm in diameter; the largest reported lesion was 4 cm. 12 Therefore, these tumors are generally found incidentally. Endoscopy demonstrates a yellowish, submucosal tumor that is usually without mucosal ulceration and has a slightly rough surface. However, it is difficult to distinguish GCTs from other submucosal tumors endoscopically. In the present study, the value of endoscopic ultrasonography was investigated. This technique is useful for evaluating submucosal tumors because their localization and origin can be visualized. Aberrant pancreas, fibromas, lipomas, and cysts can be diagnosed by analysis of echogenicity and echo pattern. 22 The ultrasonographic features of GCT have been reported for only one case previously. ~3 However, in this case and in our second case of esophageal GCT, the lesions were scanned using the balloon-contact method because it was difficult to fill the site with water. Because of tumor compression by the balloon, detailed assessVOLUME 41, NO. 2, 1995

Figure 6. Histologic appearance of large, polygonal, histiocyte-like cells with coarsely granular, amphophilic cytoplasm (H&E X200). A, Case 1. B, Case 2.

ment was not possible except for localization of the lesion. On the other hand, in our first case of gastric GCT, the ultrasonographic findings were consistent with the macroscopic findings because we did not have to use a balloon. This lesion was seen as a submucosal, homogeneous, hypoechoic mass with a clear margin and thus resembled a carcinoid tumor. Although GCTs are generally benign, especially in the gastrointestinal tract, 24 some malignant lesions have been reported. 2, 4, 25 For this reason, and also because preoperative diagnosis is difficult, the standard treatment for GCT has until now been surgical excision. Standard endoscopic biopsy is unlikely to give a correct diagnosis because the tumor is usually covered by normal mucosa and just a small quantity of tissue can be resected. Endoscopic ultrasonography may be a useful adjunctive diagnostic technique. However, it does not always allow the differential diagnosis of various submucosal tumors to be made, and it cannot necessarily distinguish benign from malignant lesions. Therefore, we used the technique of endoscopic removal by extensive biopsy in 10 patients having submucosal tumors. In all cases, the diagnosis was determined, and removal was complete in seven patients. GASTROINTESTINAL ENDOSCOPY 165

Endoscopic u l t r a s o n o g r a p h y is necessary in order to judge w h e t h e r the t u m o r m e e t s the criteria for endoscopic removal, i.e., small in size (<2 cm) a n d not att a c h e d to the muscularis propria. I f the t u m o r is initially s e p a r a t e f r o m the muscularis propria, the distance b e t w e e n t u m o r a n d muscularis p r o p r i a can be increased b y injecting solution a n d lifting the lesion, after which r e m o v a l can be carried out m o r e safely a n d completely. 26 W i t h our technique, no complications, such as bleeding or perforation, have occurred, a n d the ulcers left a f t e r r e m o v a l healed within 4 weeks in all patients. Accordingly, we consider t h a t massive h e m orrhage and p e r f o r a t i o n can be p r e v e n t e d if the m u s cularis p r o p r i a is not injured. H y p e r t o n i c saline solution with e p i n e p h r i n e ( H S E ) is injected r a t h e r t h a n n o r m a l saline solution to p r e v e n t bleeding; the vasoconstrictive action of e p i n e p h r i n e is prolonged b y hypertonic sodium. 27 I f no m a l i g n a n t change is observed in the excised s p e c i m e n after removal, we feel t h a t no additional i n t e r v e n t i o n or even follow-up is needed. In conclusion, we believe t h a t the endoscopic excision of s u b m u c o s a l t u m o r s is p r e f e r a b l e to surgical int e r v e n t i o n for cases in which the criteria specified in this r e p o r t are met. Only w h e n the excised tissue reveals findings suggestive of m a l i g n a n c y should additional, surgical i n t e r v e n t i o n be considered.

REFERENCES 1. Abrikossof AI. Weitere untersuchungen uber myoblastenmyome. Virchows Arch Pathol Anat 1931;280:723-40. 2. Strong EW, McDivitt RW, Blasfield RD. Granular cell myoblastoma. Cancer 1970;25:415-22. 3. Stefansson K, Wollmann RL. S-100 protein in granular cell tumor. Cancer 1982;49:1834-8. 4. McSwain G, Colpitts R, Kreutner A, et al. Granular cell myoblastoma. Surg Gynecol Obstet 1980;150:703-10. 5. Abdelwahab IF, Klein MJ. Granular cell tumor of the stomach. A case report and review of the literature. Am J Gastroenterol 1983;78:71-6. 6. Azzopardi JG. Histogenesis of the granular cell "myoblastoma." J Pathol 1956;71:85-95. 7. Stemmermann GN, Mori V, Waki C. Granular cell tumor (granular cell myoblastoma) of the stomach. Hawaii Med J 1961;20:537-9. 8. Goodman ML, Gottlieb LS, Zamcheck N. Granular cell myoblastoma of stomach and colon. Am J Dig Dis 1962;7:432-41. 9. Schwartz DT, Gaetz HP. Multiple granular cell myoblastomas of the stomach. Am J Clin Pathol 1965;44:453-7. 10. Murray DE, Seaman ED, Utzinger W. Granular cell myoblastomas in successive generations. J Surg Oncol 1969;1:193-7. 11. Carstens PHB. Ultrastructure of granular cell myoblastoma. Acta Pathalogica Microbiologica Scandinavica [A] 1970;78:68594. 12. Naidech HJ, Axelrod RS, Seliger G. Granular cell tumor (myoblastoma) of the stomach. AJR Am J Roentgenol 1971;113: 245-7. 13. Goodman MD, Cooper PH. Granular cell tumor (myoblastoma)

Figure 7. A, Electron micrograph in case 1 revealing the tumor cells, which contain abundant, large, autophagic granules. B, Higher magnification revealing the large granules to consist of membrane-bound, presumably autophagic vacuoles. 166 G A S T R O I N T E S T I N A L E N D O S C O P Y

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14. 15. 16. 17. 18. 19. 20. 21.

of the stomach. A case report with ultrastructural findings and review of the literature. Dig Dis 1972;17:1117-26. Aston SJ, Tompkins RK. Granular cell myoblastoma of stomach. Ann Surg 1973;177:228-31. Krouse TB, Mobini J. Multifocal granular cell myoblastoma. Report of a case involving trachea, stomach and anterior abdominal wall. Arch Pathol 1973;96:95-9. Miranda D. Benign granular cell tumor ("myoblastoma") of the stomach. Am J Gastroenterol 1976;65:344-8. Ross JS. Massive upper gastrointestinal hemorrhage from a granular cell tumor of the stomach. Am J Gastroenterol 1977; 68:595-8. Golinger RC. Granular cell myoblastoma [Letter]. JAMA 1978; 239:1493. Ghazi A. Combined granular cell tumor of the stomach and cecum. Mt Sinai J Med 1979;46:195-8. Fried KS, Arden JL, Gouge TH, et al. Multifocal granular cell tumors of the gastrointestinal tract. Am J Gastroenterol 1984; 79:751-5. Yamaguchi K, Maeda S, Kitamura K. Granular cell tumor of the

Early adenocarcinoma arising from ectopic gastric mucosa in the cervical esophagus Atsushi Takagi, Yukio Ema, Seiichi Horii, Muneji Morishita, Osamu Miyaishi, Isamu Kino,

MD MD MD MD MD MD

A l t h o u g h e c t o p i c g a s t r i c m u c o s a is o f t e n m e n t i o n e d a s a s o u r c e o f a d e n o c a r c i n o m a in t h e u p p e r e s o p h a g u s , o n l y a few c a s e s of t h i s t y p e h a v e b e e n d o c u m e n t e d . 1-6 I n t h e r e p o r t e d cases, a d e n o c a r c i n o m a s w e r e p r e s e n t beneath or adjacent to ectopic gastric mucosa, and as most of them were advanced malignancies, the exact o r i g i n c o u l d n o t b e c l e a r l y d e f i n e d . H e r e we r e p o r t a c l e a r c a s e o f a d e n o c a r c i n o m a in a n e a r l y p h a s e defin i t e l y a r i s i n g f r o m e c t o p i c g a s t r i c m u c o s a in t h e c e r vical esophagus.

CASE REPORT The patient was a 70-year-old man who first noticed severe dysphagia in December 1989. He had smoked about 10 cigarettes a day and d r u n k about 140 g of alcohol a day for about 50 years. He h a d a history of bronchial a s t h m a beginning 14 years earlier. He had undergone extensive gastrectomy at 62 years of age because of a hemorrhagic gastric ulcer.

From Kyoritsu General Hospital, Nagoya, Japan, Institute for Medical Science of Aging, Aichi Medical University, Nagakute Aichi Japan, and First Department of Pathology, Hamamatsu University School of Medicine, Hamamatsu, Japan. Reprint requests: Atsushi Takagi, MD, Kyoritsu General Hospital, 3-18-5 Rokuban, Atsuta-ku, Nagoya, 456 Japan. 0016-5107/95/4102-016753.00+ 0 GASTROINTESTINAL ENDOSCOPY Copyright 9 1995 by the American Society for Gastrointestinal Endoscopy 37/4/55873

VOLUME 41, NO. 2, 1995

22. 23. 24. 25. 26. 27.

stomach coincident with two early gastric carcinomas. Am J Gastroenterol 1989;84:656-9. Yasuda K, Nakajima M, Kawai K. Endoscopic ultrasonography in the diagnosis of submucosal tumor of the upper digestive tract. Scand J Gastroenterol 1986;21(suppl 123):59-67. Tada S, Iida M, Yao T, et al. Granular cell tumor of the esophagus: endoscopic ultrasonographic demonstration and endoscopic removal. Am J Gastroenterol 1990;85:1507-11. Lewin KJ, Riddell RH, Weinstein WM. Gastrointestinal pathology and its clinical implications. New York: Igaku-shoin, 1992. Gamboa LG. Malignant granular cell myoblastoma. Arch Pathol 1955;60:663-8. Yasuda I, Nakazawa S, Yoshino J, et al. Indication for endoscopic resection of submucosal tumor by endoscopic ultrasonography. GastroenterologicalEndoscopy (Jpn) 1993;35:240i-7. Hirao M, Kobayashi T, Masuda K, et al. Endoscopic local injection of hypertonic saline-epinephrine solution to arrest hemorrhage from the upper gastrointestinal tract. Gastrointest Endosc 1985;31:313-7.

Esophagoscopy in F e b r u a r y 1990 revealed an elevated tumor with a smooth surface and well-defined margin located 20 cm distal to the incisors (Fig. 1). A biopsy specimen of this t u m o r showed t h a t it was a well-differentiated adenocarcinoma. A barium esophagogram showed a tumorous lesion on the right wall of the cervical esophagus near the thoracic inlet (Fig. 2). In March 1990, t h e p a t i e n t underwent esophagectomy with gastric pull-up. The surgical specimen consisted of a 23-cm length of esophagus. A protruding t u m o r measuring 1.9 x 1.6 cm was present at the proximal end of the specimen (Figs. 3 and 4). A yellowish brown oval patch measuring 12 • 5 m m was situated proximal to the tumor. The distal mucosa was normal. The l y m p h nodes were not resected. Microscopic examination revealed a well-differentiated t u b u l a r adenocarcinoma composed of cells with hyperchromatic nuclei and eosinophilic cytoplasm. T h e adenocarcin o m a was observed to be contiguous with the ectopic gastric mucosa, which contained mucous glands and parietal and chief cells (Figs. 5 and 6). T h e cancer was limited almost entirely to the mucosa, with slight focal invasion to the superficial submucosa (Fig. 7). No invasion of cancer cells into lymphatic tissue or veins in the submucosa was noted. T h e a t y p i a of the t u m o r tended to increase toward the center of the tumor (Fig. 8).

DISCUSSION Adenocarcinoma of the esophagus may arise from esophageal cardiac glands, islets of ectopic gastric m u c o s a , o r t i s s u e a f f e c t e d b y B a r r e t t ' s s y n d r o m e . 1-9 I n contrast, adenoacanthoma, mucoepidermoid carcinoma, and adenoid cystic carcinoma may arise from esophageal glands. 2 Islets of ectopic gastric mucosa are found as inconstant patches apart from the stomach, most often in the proximal esophagus. They are observed most freq u e n t l y in t h e l a t e r a l e s o p h a g e a l w a l l s b e t w e e n t h e l e v e l o f t h e c a r t i l a g e a n d f i f t h t r a c h e a l ring. 3 E n d o s c o p i c a l l y , t h e y a r e s e e n as s h a r p l y d e m a r c a t e d , y e l lowish brown, oval patches. Some are very tiny and GASTROINTESTINAL ENDOSCOPY 1 6 7