included in the differential diagnosis of patients who are seenwith unusual cholangiographic findings and cholestasis.

DISCLOSURE

All authors disclosed no financial relationships rele-vant to this publication.

Abbreviations: CBD, common bile duct; LFT, liver function test; MPA,

microscopic polyarteritis; p-ANCA, perinuclear antineutrophil cytoplasm

antibodies; PAN, polyarteritis nodosa.

REFERENCES

1. Batts KP. Ischemic cholangitis. Mayo Clin Proc 1998;73:380-5.

2. Barquist ES, Goldstein N, Zinner MJ. Polyarteritis nodosa presenting as

a biliary stricture. Surgery 1991;109:16-9.

3. Kasper HU, Dries V, Drebber U, et al. Florid ischemic cholangitis due to

leucocytoclastic vasculitis. J Gastroenterol 2004;39:188-91.

4. Viola S, Meyer M, Fabre M, et al. Ischemic necrosis of bile ducts compli-

cating Schonlein-Henoch purpura. Gastroenterology 1999;117:211-4.

5. Jaeger C, Mayer G, Henrich R, et al. Secondary sclerosing cholangitis

after long-term treatment in an intensive care unit: clinical presenta-

tion, endoscopic findings, treatment, and follow-up. Endoscopy 2006;

38:730-4.

Current affiliations: Department of Gastroenterology and Hepatology,

Westmead Hospital, Sydney, Australia.

Reprint requests: Michael Bourke, MBBS, Department of Gastroenterology

and Hepatology, Westmead Hospital, Sydney West Area Health Service,

Sydney, NSW 2145 Australia.

Copyright ª 2009 by the American Society for Gastrointestinal Endoscopy

0016-5107/$36.00

doi:10.1016/j.gie.2008.03.1125

Brief Reports

Gastric-duplication cyst with an aberrant pancreatic-ductal system:an unusual cause of recurrent abdominal pain

Trupti Shinde, MD, Jennifer Lindner, DO, Jan Silverman, MD, Rad Agrawal, MD,Manish Dhawan, MD

Pittsburgh, Pennsylvania, USA

Gastric duplication cysts (GDCs) are very rare in adults.Reported here is a case of a GDC communicating with anaccessory pancreas via an aberrant duct in a 49-year-oldwoman with recurrent abdominal pain.

CASE REPORT

A 49-year-old woman was seen at an outside hospital withepigastric pain. An EGD revealed an antral mass. On a CTofthe abdomen, a cystic lesion of the antrum was seen. Shewas referred to our institution. Another EGD revealed anantral submucosal mass with a central umbilication(Fig. 1). EUS revealed a 37-mm � 26-mm submucosal cystsuggestive of GDC (Fig. 2). However, results from theaspiration of the cyst revealed a high amylase level. Hersymptoms subsided but recurred 4 months later. EUS con-firmed cyst reaccumulation, and aspiration again showeda high amylase level. No malignant cells were seen. Afteraspiration, her symptoms improved again. Because of herrecurrent symptoms, an endoscopic retrograde pancrea-tography (ERP) was performed, which revealed a secondpancreatic-duct system that originated from the main pan-creatic duct near the tail of the pancreas (Fig. 3). This sec-ond system extended parallel to the main pancreatic ductbefore attaching to the antral cyst. The patient went for sur-gery, and the accessory pancreas, aberrant pancreatic duct,

and cystic portion of the antrum were resected. The surgicalspecimen showed a 5-cm round structure identified asa cyst, which was attached to a 17-cm-long accessory pan-creas with the aberrant ductal system (Fig. 4). The cystwas unilocular, with smooth lining. On microscopic exami-nation, the cyst wall was lined by gastric mucosa with focal

Figure 1. An upper endoscopy image, showing an antral submucosal

mass with a central umbilication.

www.giejournal.org Volume 69, No. 2 : 2009 GASTROINTESTINAL ENDOSCOPY 377

Brief Reports

ulceration and a well-circumscribed smooth-musclelayer (Fig. 5). The patient’s symptoms resolved completelyafter surgical resection.

DISCUSSION

Gastric duplication cysts make up only 3.8% of all dupli-cations of the alimentary tract.1 Furthermore, GDC with an

Figure 2. EUS, revealing a cyst opening in the antrum of the stomach.

Figure 3. ERP image, revealing an aberrant pancreatic-duct system

originating from the main pancreatic duct near the tail of the pancreas, ex-

tending parallel to the main pancreatic duct, and attaching to the antral

cyst. Main pancreatic duct (a), aberrant pancreatic duct (b), antral cyst (c).

378 GASTROINTESTINAL ENDOSCOPY Volume 69, No. 2 : 2009

aberrant pancreatic duct and accessory pancreas is ex-tremely rare.1 Abnormal foregut development is believedto be responsible for this congenital anomaly.2 Severaltheories are proposed but two of them, Bremer’s theoryof errors on recanalization and McLetchie’s theory, aremost widely accepted.1 McLetchie proposed a neuroenterichypothesis in which an embryonic entoectodermal adhe-sion gives rise to a ‘‘neuroenteric’’ band, which may formtraction diverticula and lead to gut cyst formation.1,2 Thistheory explains the congenital anomaly in our patient,with the traction on the pancreatic bud by a neuroentericband causing both gastric and pancreatic abnormalities.

There are specific characteristics to define a gastric dupli-cation cyst. The cyst wall should be contiguous with the

Figure 4. Surgical specimen, showing stomach, duplication cyst, and an

aberrant pancreas.

Figure 5. Photomicrograph of cyst wall, demonstrating 4 layers: mucosa,

muscularis mucosae, submucosa, and muscularis propria (H&E, orig.

mag. �40).

www.giejournal.org

Brief Reports

stomach. It should be surrounded by a coat of sharedsmooth muscle. A common blood supply should be pres-ent. It should be lined by alimentary-tract epithelium.Rarely, respiratory-tract epithelium, in conjunction with ali-mentary-tract epithelium, has been noted.3,4

In adults, diagnosis is very difficult before surgery. Gastricduplication cysts are usually asymptomatic.5,6 However,signs and symptoms may include epigastric pain, abdominalmass, or vomiting. Rarely, GI bleeding, pancreatitis, peritoni-tis, malignancy, or even acute abdomen may result.6-8 Often,a GDC may be mistaken as a pancreatic pseudocyst, which ismore common.8 Normal levels of pancreatic enzymes andno personal history of pancreatitis may suggest a gastricduplication cyst. Unlike GDC, the pancreatic pseudocyst isgenerally associated with repeated attacks of pancreatitis.They lack an epithelial lining and contain amylase-rich fluid.8

US, CT, and EUS may help in the diagnosis.In our case, the diagnosis was made before surgery by

EUS-guided cyst aspiration and ERP. Surgical resection ledto a satisfactory outcome. This case provides a fascinatingopportunity to explore embryogenesis of the alimentarytract, as well as highlighting the usefulness of EUS andERP in preoperative diagnosis.

DISCLOSURE

All authors disclosed no financial relationships rele-vant to this publication.

Abbreviations: ERP, endoscopic retrograde pancreatography; GDCs,

gastric duplication cysts.

REFERENCES

1. Muraoka A, Tsuruno M, Katsuno G, et al. A gastric duplication cyst with

an aberrant pancreatic ductal system: report of a case. Surg Today

2002;32:531-5.

2. Camoglio FS, Forestieri C, Zanatta C, et al. Complete pancreatic ectopia

in a gastric duplication cyst: a case report and review of the literature.

Eur J Pediatr Surg 2004;14:60-2.

3. O’Donnell PL, Morrow JB, Fitzgerald TL. Adult gastric duplication cysts:

a case report and review of literature. Am Surg 2005;71:522-5.

4. Takahara T, Torigoe T, Haga H, et al. Gastric duplication cyst: evaluation

by endoscopic ultrasonography and magnetic resonance imaging.

J Gastroenterol 1996;31:420-4.

5. Johnstone DW, Forde KA, Markowitz D, et al. Gastric duplication cyst

communicating with the pancreatic duct: a rare cause of recurrent

abdominal pain. Surgery 1991;109:97-100.

6. Coit DG, Mies C. Adenocarcinoma arising within a gastric duplication

cyst. J Surg Oncol 1992;50:274-7.

7. Horne G, Ming-Lum C, Kirkpatrick AW, et al. High-grade neuroendocrine

carcinoma arising in a gastric duplication cyst: a case report with liter-

ature review. Int J Surg Pathol 2007;15:187-91.

8. Yang MC, Duh YC, Lai H, et al. Alimentary tract duplications. J Formos

Med Assoc 1996;95:406-9.

Current affiliations: Departments of Medicine (T.S.), Pathology (J.L., J.S.),

Gastroenterology (R.A. M.D.), Allegheny General Hospital,

Pittsburgh, Pennsylvania, USA.

Reprint requests: Trupti Shinde, MD, Internal Medicine, Allegheny General

Hospital, Medicine Department, 320 E. North Ave, Pittsburgh, PA 15212.

Copyright ª 2009 by the American Society for Gastrointestinal Endoscopy

0016-5107/$36.00

doi:10.1016/j.gie.2008.04.059

Pancreatic somatostatinoma and tuberous sclerosis: case report ofan exceedingly rare association

Jayaprakash Sreenarasimhaiah, MD, Luis A. Armstrong, MD, Shou-Jiang Tang, MD,Carlton Barnett, MD

Dallas, Texas, USA

Tuberous sclerosis is an autosomal dominant conditioncharacterized by the triad of mental deficiency, epilepsy,and the classic skin eruption known as adenoma seba-ceum.1 Other findings include visceral lesions with histol-ogy similar to the skin lesions, renal angiomyolipomas,and pancreatic abnormalities. Findings in the pancreascan include pancreatic cysts, rare angiomyolipomas, andneuroendocrine tumors of the nonfunctioning islet cellvariety.2 Results of certain reports suggest that functionaland nonfunctional insulinomas may be associated withthis disorder. To our knowledge, this is the first reported

case of pancreatic somatostatinoma associated with tuber-ous sclerosis.

CASE REPORT

A 24-year-old man with a history of tuberous sclerosiscomplained of 3 months of dull abdominal pain. He deniedsymptoms of GI bleeding, jaundice, nausea, or vomiting, orassociation with food intake. He reported a mild weight lossof 10 pounds during this period. His history also included

www.giejournal.org Volume 69, No. 2 : 2009 GASTROINTESTINAL ENDOSCOPY 379