Clin J Gastroenterol (2013) 6:156–159 DOI 10.1007/s12328-013-0367-0

CASE REPORT

Intrapancreatic gastric duplication cyst mimicking pancreatic cystic tumor Keiko Kamei • Takeo Yasuda • Shumpei Satoi • Hajime Ishikawa • Hiroki Sakamoto • Masayuki Kitano • Takaaki Chikugo • Takuya Nakai • Yoshifumi Takeyama

Received: 30 October 2012 / Accepted: 23 January 2013 / Published online: 2 March 2013 Ó Springer Japan 2013

Abstract Cystic lesions of the pancreas are sometimes difficult to diagnose. We report a case of a gastric duplication cyst (GDC) of the pancreas in an adult. A 45-year-old woman was admitted to our department for the investigation of anemia. Abdominal ultrasonography revealed a large cystic lesion, measuring about 40 9 70 mm with calcification in the tail of the pancreas. Contrast-enhanced computed tomography of the abdomen revealed a non-enhanced cystic lesion with non-enhanced wall. Endoscopic retrograde pancreatography revealed a mild extended main pancreatic duct and an aberrant pancreatic duct, but there was no communication with the cyst. We could not deny the malignancy, so distal pancreatectomy was performed. The pathological examination revealed that the inner wall of the cyst consisted of columnar epithelium and smooth muscle layer. Immunohistochemical analysis revealed the columnar epithelium to be immunopositive for cytokeratin 7 (CK7) and immunonegative for cytokeratin 20 (CK20) and the glands to be immunonegative for CK 7 and immunopositive for CK 20. Therefore, a diagnosis of GDC of the pancreas was made. On imaging, a GDC usually mimics a pancreatic pseudocyst

or a cystic neoplasm of the pancreas. Therefore, even though it is rare, a GDC should be considered in the differential diagnosis of cystic tumors of the pancreas. Keywords Gastric duplication cyst  Pancreas  Aberrant pancreatic duct system  Distal pancreatectomy

Introduction Duplication of the gastrointestinal tract can occur anywhere from the mouth to the anus. Among duplication of the gastrointestinal tract, gastric duplication cysts (GDCs) comprise 4 % of all alimentary duplications [1]. Most of these anomalies are diagnosed in childhood, and GDC in the pancreas is especially rare. It usually mimics a pancreatic pseudocyst or a cystic tumor, and is difficult to diagnose. Here we report a case of a GDC in the pancreas of a 45-year-old woman and we review the literature.

Case report K. Kamei  T. Yasuda (&)  S. Satoi  H. Ishikawa  T. Nakai  Y. Takeyama Department of Surgery, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-sayama 589-8511, Japan e-mail: [email protected] H. Sakamoto  M. Kitano Department of Gastroenterology and Hepatology, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-sayama 589-8511, Japan T. Chikugo Department of Pathology, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-sayama 589-8511, Japan

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A 45-year-old woman was referred to our hospital for investigation of anemia found during a routine medical check-up. She was asymptomatic and had no medical history including pancreatitis. Laboratory tests showed moderate anemia (hemoglobin 9.5 g/dl, hematocrit 32.3 %). Upper gastrointestinal endoscopy and colonoscopy revealed no abnormal findings. Abdominal computed tomography (CT) demonstrated a cystic lesion which was not enhanced with contrast material at the tail of the pancreas (Fig. 1a, b). Endoscopic ultrasonography also revealed a cystic lesion with calcification inside. Endoscopic retrograde pancreatography revealed a mild extended main pancreatic duct and

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the stomach. The resected specimen revealed that the cyst existed in the pancreas tail, and that it contained white viscid matter (Fig. 3a, b). There was no nodule or tumor inside the cyst (Fig. 3c). A cross-section of the resected specimen confirmed that the cyst was present in the pancreas and cyst (Fig. 4a). The pathological examination of this cystic lesion revealed that the inner wall of the cyst consisted of columnar epithelium and smooth muscle layer with a small amount of ganglion cells (Fig. 4b, c). The glands of this cyst consisted of cells with eosinophil granules and cells with basophil granules, suggesting that they were gastric fundic glands. This cyst had no communication with the pancreatic duct. Immunohistochemical analysis revealed the columnar epithelium to be immunopositive for cytokeratin 7 (CK7) and immunonegative for cytokeratin 20 (CK20) and the glands to be immunonegative for CK7 and immunopositive for CK20. Moreover, the glands were immunonegative for MUC1 and MUC2, and immunopositive for MUC5AC and MUC6. Thus, a final diagnosis of intrapancreatic GDC was made. The postoperative course was uneventful and the patient is doing well after the surgery without any sign.

Discussion

Fig. 1 a Abdominal computed tomography (axial view) demonstrated a cystic lesion which was not enhanced with contrast material at the tail of the pancreas. Cystic lesion contained calcification material. b Abdominal computed tomography (coronal view) demonstrated a cystic lesion near the stomach

Fig. 2 Endoscopic retrograde pancreatography revealed a mild extended main pancreatic duct and an aberrant pancreatic duct. There was no communication between the pancreatic duct system and the cystic lesion

an aberrant pancreatic duct, but no communication with the cystic lesion was detected (Fig. 2). As we could not deny malignancy, we performed a distal pancreatectomy. At surgery, a 70-mm maximum diameter cyst was found at the pancreatic tail, which was free from other organs including

Duplication of the gastrointestinal tract can occur anywhere from the mouth to the anus. They are spherical or tubular structures that are covered inside by gastrointestinal mucosa containing a distinct layer of smooth muscle [2]. The reason why duplication of the gastrointestinal tract occurs is partly explained by the report of Lewis and Thyng in 1908 [3]. They studied the embryos of pigs, rabbits and man, and reported the regular occurrence of nodules of epithelial cells that become vacuolated and break off the alimentary tract or remain in continuity to from diverticula. Normally, these diverticula regress, but occasionally they persist, giving rise to cysts. Among duplication of the gastrointestinal tract, GDCs comprise 4 % of all alimentary duplications [1]. The most common location of GDCs is along the greater curvature, mostly in the antrum [4], and an intrapancreatic GDC, like our case, is extremely rare. Ladd et al. [5] reported the essential features of GDCs as (1) the cyst wall is contiguous with the stomach; (2) it is surrounded by a smooth muscle, which fuses with the muscle layer of the stomach; and (3) it is lined with alimentary epithelium. However, some authors reported GDC that was not contiguous with the stomach like our case [6, 7]. Recently, Kohno et al. [8] classified GDCs into 4 categories¯those without any communication with the pancreatic duct, noncontiguous with the stomach (category 1); those without communication with the pancreatic duct, contiguous with the stomach (category 2); those with

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Fig. 3 a The resected specimen revealed that the cyst existed in the pancreas tail. b Cystic tumor contained white viscid matter. c There was no nodule or tumor inside the cyst

Fig. 4 a Cross-section of the resected specimen confirmed the adherence of the pancreas and cyst. b, c The pathological examination (H&E stain) of this cystic lesion revealed that the inner wall of the

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cyst consisted of columnar epithelium and smooth muscle layer. b Low-power field, c high-power field

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communication with the pancreatic duct, noncontiguous with the stomach (category 3); and those with communication with the pancreatic duct, contiguous with the stomach (category 4). Our case falls into category 1. The symptoms of GDCs are usually nonspecific, depending on the location. Common symptoms include recurrent abdominal pain, caused by hemorrhaging within the cysts, inflammation, ulcers due to gastric acid secretion from the mucosa, or pancreatitis. Gastrointestinal perforation and bleeding have also been reported [9, 10]. Moreover, Muraoka et al. [11] reviewed 11 cases of GDC with an aberrant pancreatic ductal system, and reported that such patients had some abdominal pain related to pancreatitis. In our case, although the patient had GDC with an aberrant pancreatic ductal system, the patient had no abdominal pain or medical history related to GDC and pancreatitis. GDCs without any symptoms are usual, but GDCs with an aberrant pancreatic ductal system revealing no symptoms are rare; this makes a precise preoperative diagnosis difficult, as in our case. Diagnosis is usually made early in life; approximately 70 % of patients present within the first year after birth. As it is a rare entity in adults [4], it is difficult to obtain a preoperative diagnosis of GDC in adults. Contrastenhanced CT demonstrates GDC as a thick-walled cystic lesion with enhancement of the inner lining [12, 13]. Calcification is occasionally observed on CT [14]. These findings are of diagnostic significance for GDCs; however, CT scans may sometimes fail to differentiate a GDC from a pseudocyst or cystic tumor of the pancreas, as in our patient. If a gastric duplication is suspected preoperatively, which is rare, technetium-99m imaging will identify duplications containing gastric mucosa. Eleven cases of GDC with an aberrant pancreatic ductal system, as mentioned above, were summarized by Muraoka et al. since Bradbeer [6] described the first case in 1959. Nine of these cases (75 %), including our case, were female with a mean age at the operation of 20.4 years (range 3-46 years). The main symptoms were abdominal pain related to pancreatitis (11 cases, 92 %). In addition, 4 cases including our case (33 %) were not contiguous with stomach, 3 cases including our case (25 %) had no communication with pancreatic duct, and only 1 case (8 %) had communication with gastric lumen. The duplication cyst should be excised because it sometimes has been the cause of morbidity; even adenocarcinoma has been reported in foregut duplications [15]. The optimal treatment procedure varies depending on its location in the pancreas and the relationship with the pancreatic duct. Enucleation of GDC is ideal to prevent the development of pancreatic insufficiency in patients, but

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pancreatic resections are often performed. Nowadays, laparoscopic procedure is applied to this disease [11]. In our case, a malignant tumor could not be ruled out, so a distal pancreatectomy with splenectomy was performed. In retrospect, a spleen-preserving distal pancreatectomy was ideal. Finally, we conclude that, although they are rare, GDCs should be considered in the differential diagnosis of cystic tumors of the pancreas even in adults. Conflict of interest interest.

We declare that they have no conflict of

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Intrapancreatic gastric duplication cyst mimicking pancreatic cystic tumor.

Cystic lesions of the pancreas are sometimes difficult to diagnose. We report a case of a gastric duplication cyst (GDC) of the pancreas in an adult. ...
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