287Copyrights © 2015 The Korean Society of Radiology

INTRODUCTION

Gastrointestinal stromal tumor (GIST) is one of the most common mesenchymal tumors of the gastrointestinal tract and most commonly located in the stomach (1). Bleeding and ane-mia are the common presentations of GISTs and larger tumors may also present with pain and obstruction (2, 3). However, GISTs rarely cause hemoperitoneum. In this article, we reported a case of a large pedunculated GIST causing loculated hemato-ma within the gastrocolic ligament.

CASE REPORT

A 52-year-old man presented with a 3-week history of abdomi-nal distension and epigastric pain. At admission, the patient com-

plained of aggravated squeezing epigastric pain and severe nau-sea. His vital signs were stable, but hemoglobin level decreased from 16.5 g/dL (5 days prior) to 12.0 g/dL, and C-reactive protein was elevated to 3.10 mg/dL. Complete blood count, electrolytes, liver function tests, and urinalysis were normal. He had no history of abdominal trauma or surgery, and no significant weight loss. On physical examination, he complained of tenderness in the epi-gastrium during palpation, but rebound tenderness was absent.

Computed tomography (CT) (LightSpeed16, GE Healthcare, Milwaukee, WI, USA) showed a large, well-defined heteroge-neously enhancing mass containing central necrotic area of low attenuation in the mid abdomen (Fig. 1A). The mass measured 10.3 × 7.5 × 9.4 cm. The mass was surrounded by peritumoral hematoma within the gastrocolic ligament and small amount of hemoperitoneum was identified in the left inframesocolic space.

Case ReportpISSN 1738-2637 / eISSN 2288-2928J Korean Soc Radiol 2015;72(4):287-290http://dx.doi.org/10.3348/jksr.2015.72.4.287

Received August 15, 2014; Accepted January 29, 2015Corresponding author: Sung Eun Ahn, MDDepartment of Radiology, Kyung Hee University Medical Center, Kyung Hee University School of Medicine, 23 Kyungheedae-ro, Dongdaemun-gu, Seoul 130-872, Korea.Tel. 82-2-958-8622 Fax. 82-2-968-0787E-mail: 83ahnse@hanmail.net

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distri-bution, and reproduction in any medium, provided the original work is properly cited.

Gastric gastrointestinal stromal tumor (GIST) is one of the most common mesenchy-mal tumors of the stomach, which may be asymptomatic or cause symptoms such as pain, gastrointestinal bleeding, and obstruction. Hemoperitoneum due to spontaneous rupture of the tumor is an extremely rare complication. We described a case of a 52-year-old man with a large pedunculated GIST causing loculated hematoma within the gastrocolic ligament. The patient visited our hospital due to a 3 week history of epigastric pain. A computed tomography scan revealed a 10.3 × 7.5 × 9.4 cm sized mass that was growing exophytically from the greater curvature of the stomach and was surrounded by loculated hematoma within the gastrocolic ligament. Laparotomy revealed a large stalked gastric mass surrounded by loculated hematoma within the gastrocolic ligament and blood fluid in the peritoneal cavity. Pathologic examination confirmed a GIST, of the high risk group.

Index termsGastrointestinal Stromal TumorHematoma, Stomach

Spontaneous Rupture of Pedunculated Gastric Gastrointestinal Stromal Tumor into the Gastrocolic Ligament Presenting as a Stalked Mass Surrounded by Loculated Hematoma1

위결장인대 내로 자발 파열된 유경성 위 위장관 간질종양1

Hyun Soo Kim, MD1, Sung Eun Ahn, MD1, Seong Jin Park, MD1, Sung Kyoung Moon, MD1, Joo Won Lim, MD1, Dong Ho Lee, MD1, Yong Ho Kim, MD2

Departments of 1Radiology, 2Surgery, Kyung Hee University Medical Center, Kyung Hee University School of Medicine, Seoul, Korea

Spontaneous Rupture of Pedunculated Gastric GIST into the Gastrocolic Ligament

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peritoneum and the abdomen was irrigated with saline. The mass showed adhesions with the adjacent omentum, and adhe-siolysis following wedge resection of mass was conducted.

On macroscopic inspection, we found a round lobulated mass that was focally attached to the gastric wall with stalk. The mass measured 13 × 10 × 9 cm in size and had a stalk of 2 cm in length. The cut surface of the lobulated mass showed cystic spaces, hemorrhage, and gray parenchyma with myxoid change (Fig. 1E). On histological examination, the tumor was found to invade the muscularis propria of the stomach and the mass was composed of spindle-shaped cells. Up to 3 mitotic figures were counted in 50 high-power fields.

Immunohistochemistry showed that the tumor was c-KIT positive. The tumor was negative for CD34, desmin, and S100.

The mass was connected to the greater curvature of the stomach by a stalk that contains the vessels of gastric origin supplying the mass (Fig. 1B, C). The mass did not show extension to the mu-cosal layer of the stomach. These CT findings suggested a pe-dunculated, subepithelial tumor growing exophytically from the greater curvature of the stomach with a stalk, and loculated he-matoma within the gastrocolic ligament with hemoperitoneum due to the ruptured mass.

The patient underwent diagnostic laparoscopy after radiologi-cal examination. Laparoscopy revealed a large mass surrounded by hematoma in the greater curvature side of the posterior wall of the stomach (Fig. 1D). The mass was hemorrhagic. The pro-cedure was converted to an upper midline laparotomy for mass and hematoma removal. The patient had about 2 liters hemo-

Fig. 1. A 52-year-old man with a large pedunculated gastric gastrointestinal stromal tumor causing loculated hematoma within the gastrocolic ligament.A. The axial image of the contrast-enhanced abdominal CT scan reveals a heterogenous mass with a central area of low attenuation in the mid abdomen (arrow). There is peritumoral hematoma within the gastrocolic ligament (arrowhead) and loculated hemoperitoneum in the left in-framesocolic space (asterisk).B. The coronal image demonstrates a stalk between the greater curvature of the stomach and the mass, which indicates that the mass is growing exophytically from the greater curvature of the stomach and the stalk contains the arteries supplying the mass (arrow).C. The sagittal image clearly demonstrates a stalk, which contains the gastric arteries (arrow). D. Laparoscopy reveals a large, mass surrounded by hematoma (asterisk) in the greater curvature side of posterior wall of the stomach. The blood was oozing from the mass.E. Macroscopic image of the surgical specimen after resection of the mass. The tip of the mass shows the stalk (asterisk) attached to gastric wall. The cut surface of the lobulated mass shows hemorrhage (arrows) and myxoid degeneration (square).

D

A

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Hyun Soo Kim, et al

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the reviewed cases showed an exophytic growth pattern (12 of 13 cases). Especially, 5 cases showed an exophytic mass that was focally attached to the gastric wall with a pedunculated stalk. This case also showed an approximately 10 cm sized, large, pe-dunculated mass, growing exophytically from the greater curva-ture of the stomach. Larger tumors are possibly more vulnerable to rupture because of intratumoral necrosis. In addition, exo-phytic growth pattern with a pedunculated stalk might increase the risk of tumor rupture because the exophytic growing mass with a pedunculated stalk has higher mobility with increased risk for adjacent omental or mesenteric torsion or strangulation and complete obstruction of blood supply. In our case, the mass showed an island-like appearance, and the heterogeneously en-hancing mass was surrounded by hematoma appearing as high attenuation fluid on CT examination.

Features that are associated with a poor prognosis include large tumor size, cell type (epithelioid versus spindle), cellularity, mitotic count, necrosis, and the presence of metastases (6-8). Tumor rupture (either spontaneous or iatrogenic) reportedly in-creases the risk of tumor recurrence due to peritoneal seeding (9, 10). In our case, the postoperative CT scan performed at 3 months after surgery showed no evidence of tumor recurrence. Regular patient follow-up was scheduled.

In conclusion, although hemoperitoneum due to spontaneous GIST rupture is a rare condition, GIST should be considered in the differential diagnosis of spontaneous intra-abdominal bleed-ing, especially if the mass shows an exophytic growth pattern and has a pedunculated stalk.

Overall assessment confirmed the diagnosis of GIST, of the high risk group.

DISCUSSION

GIST is one of the most common mesenchymal tumors of the gastrointestinal tract. The most common anatomical sites of ori-gin are the stomach (40–60%), small intestine (30–40%), colon and rectum (5%) (1). Clinical presentation of gastric GIST de-pends on the tumor size and location. GIST is usually asymp-tomatic, as long as it is relatively small (2). Bleeding and anemia are the common presentations (42%), and larger tumors may also present with pain and obstruction (3, 4).

However, hemoperitoneum due to GIST rupture is a rare con-dition. The presence and extent of hemoperitoneum can be identified by CT, and it may appear as hyperdense ascites. The reason for the occurrence of hemoperitoneum from spontane-ous rupture of GISTs is unclear. Hematoma formation within the tumor, and the resultant free hemorrhage by rupture of a weakened area in the tumor wall may be due to extensive intra-tumoral necrosis (3-5).

PubMed search of the English literature identified around 13 cases of ruptured gastric GISTs causing hemoperitoneum with imaging studies. These cases were summarized in Table 1, in-cluding patient characteristics such as age, sex, symptoms, imag-ing findings (size and growth pattern) and prognostic group. The mean size of the reviewed ruptured gastric GISTs causing hemoperitoneum was 7.8 cm (range, 1.3–11 cm). Also, most of

Table 1. Summary of the 13 Cases of Ruptured Gastric GIST Causing HemoperitoneumAuthor Age/Sex Symptoms Size (cm) Growth Pattern Prognostic Group

1 Cho 71/F Abdominal pain 6 × 8 Exophytic broad based High risk 2 Cheon 38/M Abdominal pain 10 × 9 Exophytic focally attached Intermediate 3 Cegarra 83/F Abdominal pain, anemia 7.5 Exophytic High risk 4 Cegarra 79/F Abdominal pain, anemia 11 Exophytic Intermediate 5 Park 47/F Abdominal pain, anemia 7 Exophytic broad based High risk 6 Bucher 49/M Abdominal pain, severe hypotension 10 Exophytic High risk 7 Seya 60/M Syncope, hematemesis 6 Exophytic Intermediate 8 Bae 33/M Syncope 4 Exophytic Intermediate 9 Fiscon 68/M Abdominal pain 10 × 7 Exophytic focally attached High risk10 Mohamed 40/M Abdominal pain, minor trauma 10 × 9 Exophytic stalk Intermediate11 Benjamin 54/M Abdominal pain, drop in Hb 1.3 Subepithelial mass High risk12 Yakan 51/M Abdominal pain 6 × 5 Exophytic pedunculated Intermediate13 Tiffaney 17/M Abdominal pain 11 × 8 Exophytic pedunculated, twisted Intermediate

Note.—GIST = gastrointestinal stromal tumor

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7. Varras M, Vlachakos N, Akrivis C, Vasilakaki T, Skafida E.

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hemoperitoneum in puerperium: report of a case with re-

view of the literature. World J Surg Oncol 2010;8:95

8. Jacobs K, de Gheldere Ch, Vanclooster P. A ruptured gas-

trointestinal stromal tumour of the transverse mesocolon:

a case report. Acta Chir Belg 2006;106:218-221

9. Takahashi T, Nakajima K, Nishitani A, Souma Y, Hirota S,

Sawa Y, et al. An enhanced risk-group stratification system

for more practical prognostication of clinically malignant

gastrointestinal stromal tumors. Int J Clin Oncol 2007;12:

369-374

10. Rutkowski P, Nowecki ZI, Michej W, Debiec-Rychter M,

Wozniak A, Limon J, et al. Risk criteria and prognostic fac-

tors for predicting recurrences after resection of primary

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위결장인대 내로 자발 파열된 유경성 위 위장관 간질종양1

김현수1 · 안성은1 · 박성진1 · 문성경1 · 임주원1 · 이동호1 · 김용호2

위장관 간질종양은 위에서 발생하는 가장 흔한 간엽성 종양 중 하나이다. 임상적으로는 증상이 없거나 비 특이적인 복통,

위장관 출혈에 의한 증상을 보일 수 있으나, 혈복증이 생기는 경우는 매우 드물다. 저자들은 외방성 성장을 한 위장의 유

경성 위장관 간질종양이 자발 파열되어 위결장인대에 국소적 혈종을 형성한 52세 남자 환자의 증례를 보고하고자 한다.

환자는 3주 전부터 시작된 복통을 주소로 내원하였으며 이후 시행한 CT에서 10.3 × 7.5 × 9.4 cm 크기의 종괴가 위의

대만부에서 외방성으로 자라나고 있었고 주변으로 국소적인 혈종이 위결장인대 내에서 관찰되었다. 개복술을 통하여 혈종

으로 둘러싸인 유경성의 종괴가 위결장인대 내에서 발견되었으며 혈복증을 동반하고 있었고 병리학적 검사에서 고위험군

위장관 간질종양으로 확진되었다.

경희대학교 의과대학 경희대학교병원 1영상의학과학교실, 2외과학교실