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INTRODUCTION

Gastritis cystica polyposa (GCP) is a rare condition, which is histologically characterized by polypoid hyperplasia of the gas-tric mucosa with cystic dilation of glandular structures (1). GCP usually occurs at the gastroenterostomy site, almost al-ways on the gastric side of the anastomosis (2, 3). Chronic mu-cosal irritation, by bile reflux and increased mucosal mobility in the operated stomach, are thought to play important roles in the pathogenesis of GCP (3). Nevertheless, GCP is rarely found in patients without any prior history of gastric surgery (1, 4, 5). In these cases, GCP is thought to be related to chronic gastritis or ischemia (1). To the best of our knowledge, there are only a few reports of GCP arising from the unoperated stomachs. Also, there are only a few reports concerning the radiologic findings of GCP. Hence, we report the case of GCP originating in the unoperated stomach.

CASE REPORT

A 57-year-old man was referred to our hospital for further evaluation of a gastric mass that was detected by endoscopy. The patient had no significant symptoms other than melena. There was nothing unusual observed on the physical examina-tion, and routine laboratory test results were normal. He had no specific family or past medical history. On review of endo-scopic pictures from the previous hospital that the patient had visited, a polypoid mass with ulceration was found in the gas-tric antrum, and the gastric mass was covered with erythema-tous gastric mucosa (Fig. 1A). Submucosal hemorrhages were noted in the adjacent gastric mucosa. Fifty-one days after the first endoscopy, the patient underwent endoscopy for the sec-ond time. A polypoid mass, which measured 24 mm in diame-ter, was found on the greater curvature of the stomach (Fig. 1B). Erythema of gastric mucosa that covered the surface of the

Case ReportpISSN 1738-2637J Korean Soc Radiol 2012;67(4):253-256

Received April 16, 2012; Accepted July 24, 2012Corresponding author: Kyung Ah Kim, MDDepartment of Radiology, Research Institute of Radiological Science, Severance Hospital, Yonsei University College of Medicine, 50 Yonsei-ro, Seodaemun-gu, Seoul 120-752, Korea. Tel. 82-2-2228-7400 Fax. 82-2-393-3035E-mail: bellenina@yuhs.ac

Copyrights © 2012 The Korean Society of Radiology

Gastritis cystica polyposa (GCP) is an uncommon lesion that usually develops at the gastroenterostomy site. A 57-year-old man visited a hospital with a complaint of me-lena. He did not have any surgical history or past medical history. Endoscopy was per-formed to evaluate the cause of melena, and a polypoid cystic mass in the stomach was found on an endoscopy and endoscopic ultrasonography. The polypoid cystic mass did not show any enhancing solid portion on a computed tomography. The gas-tric lesion was conclusively confirmed as GCP through endoscopic submucosal dissec-tion. We report a rare case of GCP that occurred in an unoperated stomach.

Index termsGastritis Cystica PolyposaComputed TomographyEndoscopic UltrasonographyEndoscopic Submucosal Dissection

Gastritis Cystica Polyposa in the Unoperated Stomach: A Case Report1

위수술의 과거력이 없는 환자에서 발생한 낭성용종성위염: 증례 보고1

Yeo Jin Lee, MD1, Kyung Ah Kim, MD1, Hyae Min Jeon, MD2, Yong Eun Chung, MD1, Joon Seok Lim, MD1 1Department of Radiology, Research Institute of Radiological Science, 2Department of Pathology, Severance Hospital, Yonsei University College of Medicine, Seoul, Korea

Gastritis Cystica Polyposa in the Unoperated Stomach

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marcated, anechoic mass with narrow stalk in the submucosal layer of the stomach (Fig. 1C). No solid component or mural nodule was found within this cystic mass on EUS. Contrast-en-hanced computed tomography (CT) was performed, and the

polypoid mass exhibited improvement and the ulcer previously detected on the mass was nearly healed (Fig. 1B). Adjacent sub-mucosal hemorrhages were almost resolved. Endoscopic ultra-sonography (EUS) was performed and it revealed a well-de-

A B C

Fig. 1. A 57-year-old man with gastritis cystica polyposa in the unoperated stomach, which was confirmed by an endoscopic mucosal dissection. A. The initial endoscopic image shows a polypoid mass of 2.4 cm in size with mucosal ulceration and erythema on the mass. Submucosal hemor-rhage is shown in the adjacent gastric mucosa. B. Erythema of gastric mucosa and ulceration on the surface of the polypoid mass are improved on the follow-up endoscopic image. C. EUS image shows a unilocular, cystic mass in the submucosal layer without any solid component. The mass has a narrow stalk (arrows). D, E. The axial (D) and coronal (E) images of contrast-enhanced CT show a polypoid, cystic mass (arrows) with a well-defined margin on the greater curvature of the stomach. Intact gastric mucosa is seen overlying the mass. F. The gross specimen shows a polypoid mass with a submucosal stroma measuring 2 × 1.8 × 2.5 cm. Cross-sectioning of the specimen reveals a unilocular cyst with a background of pale brown mucoid submucosal stroma. G. On microscopic examination, there is a large submucosal cyst (arrowheads) lined with gastric epithelium. Some elongated gastric glands (ar-rows) penetrate the muscularis mucosa (Hematoxylin-Eosin stain, × 12.5).Note.-EUS = endoscopic ultrasonography

D

F

E

G

Yeo Jin Lee, et al

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However, some literature recently reported cases of GCP devel-oping in patients with no surgical history (2, 4, 7). To date, few-er than 10 cases have been reported, which developed in pa-tients without a prior history of stomach surgery (2, 4, 5, 7, 8). The pathogenesis of GCP development in the unoperated stomach is thought to be caused by chronic ischemia and in-flammation. Despite of various pathogenesis suggested for both the operated and the unoperated stomachs, the actual patho-genesis of GCP is not fully understood.

In most cases, GCP is incidentally found by endoscopy or CT. GCP can appear to mimic a giant gastric fold, submucosal tu-mor, or isolated polyp on endoscopy. GCP usually appears as a well-defined, polypoid mass in the submucosal layer endoscopi-cally and radiologically (1). There are only a few reports describ-ing the imaging findings of GCP in the past literature (1, 2). In 1994, Wu et al. (2) reported CT findings of GCP, a homogenous-ly enhancing polypoid lesion or heterogeneously-enhancing wall thickening, with or without small cystic components. Park et al. (7) reported a GCP case that revealed a multilocular cystic mass with a thick peripheral wall on CT. In our case, GCP was a well-defined, cystic mass without any internal solid portion observed in the submucosal layer on CT and EUS. GCP reported by To-mizuka et al. (9) was similar to our case in regard to cystic mass, but was dissimilar in that it had lobulated contours and uneven wall. Our case was also different from the case described by Wu et al. (2) because the mass in our case had no enhancing solid portion. CT and EUS can demonstrate the morphologic ap-pearance of a gastric mass lesion and reveal the cystic character of the lesion in the case of GCP (7); however, such findings are not specific for GCP. It is difficult to differentiate GCP from the other cystic lesions of the stomach because of a substantial overlap of radiologic findings among them. Many cystic lesions of benign nature can arise from the stomach, including duplica-tion cyst, cystic lymphnagioma, heterotopic pancreas, or gastritis cystica polyposa (1). Solid tumors, such as schwannoma or GIST with cystic change and mucinous adenocarcinoma, originating from the stomach can also mimic a GCP (1). It is occasionally difficult to differentiate GCP from gastric cancer. There are some reports describing the association of GCP with early or small cancerous lesions (5, 7, 8). One of the difficulties is that it is still not clear whether GCP is a precancerous lesion or not. However, if GCP is confirmed histologically, complete excision

mass appeared to be a polypoid, cystic mass with a well-defined margin on the greater curvature of the stomach on CT (Fig. 1D, E). There was no enhancing portion within the mass. Nei-ther perigastric infiltration nor lymphadenopathy was found around the mass. The cystic mass was considered to be a sub-mucosal lesion of benign nature, through EUS and CT. Lesions of benign cystic nature in gastric submucosa were considered for the differential diagnoses, such as duplication cyst, cystic lymphangioma, and pseudocyst originating from the hetero-topic pancreas. In addition, we included gastrointestinal stro-mal tumor (GIST) or schwannoma, with cystic degeneration as tumorous conditions, as another possible differential diagno-ses. Endoscopic submucosal dissection (ESD) was done, and the cystic mass was completely excised. Observed as a gross specimen, the lesion was soft with pale-brown color and its di-mensions measured were 2 × 1.8 × 2.5 cm (Fig. 1F). The mass was located in the submucosal layer and an empty space was observed within the mass (Fig. 1F). Upon microscopic exami-nation, the mass was a large submucosal cyst, lined with the gastric surface epithelium. The large submucosal cyst did not have any smooth muscle, which is needed for the diagnosis of duplication cyst. Separated from the submucosal cyst, some elongated gastric glands penetrated the muscularis mucosa (Fig. 1G). With the above observations, the cystic mass and elongated gastric glands can be conclusively diagnosed as gas-tritis cystica polyposa.

DISCUSSION

Gastritis cystica polyposa is a rare polypoid lesion, which usually occurs at the gastroenterostomy site (2, 3). It was first described by Littler and Gleibermann (6) in 1972. Several syn-onyms have been used, such as multiple polypoid cystic gastri-tis, gastric cystic polyposis, stomal polypoid hypertrophic gas-tritis, and gastritis cystica profunda (3, 7). Histologically, it is characterized by the elongation of the gastric foveolae with hy-perplasia and cystic dilatation of the gastric glands extending into the gastric submucosal layer (1). GCP is most frequently seen on the gastric side of the anastomosis (2, 3). The patho-genesis of GCP is thought to occur as the epithelial elements, after surgery, migrate into the submucosa by increased mucosal mobility and by chronic mucosal damage due to bile reflux (7).

Gastritis Cystica Polyposa in the Unoperated Stomach

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funda in a patient with no history of gastric surgery. En-

doscopy 2007;39 Suppl 1:E80-E81

5. Tuncer K, Alkanat M, Musoglu A, Aydin A. Gastritis cystica

polyposa found in an unoperated stomach: an unusual

case treated by endoscopic polypectomy. Endoscopy 2003;

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6. Littler ER, Gleibermann E. Gastritis cystica polyposa. (Gastric

mucosal prolapse at gastroenterostomy site, with cystic and

infiltrative epithelial hyperplasia). Cancer 1972;29:205-209

7. Park JS, Myung SJ, Jung HY, Yang SK, Hong WS, Kim JH, et

al. Endoscopic treatment of gastritis cystica polyposa found

in an unoperated stomach. Gastrointest Endosc 2001;54:

101-103

8. Park CH, Park JM, Jung CK, Kim DB, Kang SH, Lee SW, et al.

Early gastric cancer associated with gastritis cystica polypo-

sa in the unoperated stomach treated by endoscopic sub-

mucosal dissection. Gastrointest Endosc 2009;69:e47-e50

9. Tomizuka T, Mazaki T, Mado K, Henmi A, Ishii Y, Masuda H,

et al. A case of gastritis cystica profunda. Surgery 2008;

143:449-450

is necessary (5). When GCP has no malignant portions, ESD is the proper management method in comparison to wedge re-section or more extensive gastrectomy (7). The extent of sur-gery depends on each individual case.

This case was an unusual GCP that developed in the unoper-ated stomach. GCP in this case appeared as a polypoid, cystic mass with overlying gastric mucosa on EUS and CT. GCP would be considered as a possible differential diagnosis for a cystic mass of the stomach.

REFERENCES

1. Lee J, Park CM, Kim KA, Lee CH, Choi JW, Shin BK, et al. Cys-

tic lesions of the gastrointestinal tract: multimodality imag-

ing with pathologic correlations. Korean J Radiol 2010;

11:457-468

2. Wu MT, Pan HB, Lai PH, Chang JM, Tsai SH, Wu CW. CT of

gastritis cystica polyposa. Abdom Imaging 1994;19:8-10

3. Franzin G, Novelli P. Gastritis cystica profunda. Histopa-

thology 1981;5:535-547

4. Béchade D, Desramé J, Algayres JP. Gastritis cystica pro-

위수술의 과거력이 없는 환자에서 발생한 낭성용종성위염: 증례 보고1

이여진1 · 김경아1 · 전혜민2 · 정용은1 · 임준석1

낭성용종성위염(gastritis cystica polyposa)은 주로 위소장문합술 후에 문합부에서 발생하는 드문 병변이다. 위수술의 과

거력이 없는 57세 남자가 흑색변의 원인을 찾기 위해 시행한 위내시경에서 용종성 병변이 우연히 발견되었다. 전산화단층

촬영에서 이 병변은 조영증강되는 고형 부분이 관찰되지 않고 순수 낭성 종괴로 보였다. 이후 내시경점막절제술로 절제하

여 낭성용종성위염으로 확진된 증례를 보고한다.

연세대학교 의과대학 세브란스병원 1영상의학과, 2병리과