Image of the Month—Diagnosis

This case of a previously healthy young adult presenting with a congenital anomaly highlights several interesting aspects of this rare disease. Duplication cysts most commonly present in the patient's first 1 to 2 years of life^1,2 and can occur anywhere along the alimentary tract.^3,4 Gastric duplication cysts make up less than 10% of all gastrointestinal duplications.⁵ Rarer still is the occurrence of this type of cyst in adults,⁶ which can make the diagnosis particularly challenging. In this case, the differential diagnosis prior to endoscopy included various disorders that can cause partial or complete gastric outlet obstruction, including other gastrointestinal duplications, choledochal cyst, mesenteric cyst, cystic pancreatic mass, and cystic tumors.

Various surgical approaches to removing gastric duplication cysts have been described. Extraluminal gastric duplication cysts may be resected using conventional laparoscopy.⁷ Combined endoscopy/laparoscopy has been reported for a case near the gastroesophageal junction.⁸ For smaller intraluminal lesions, an endoscopic approach with snare resection may be performed.⁹ In our case, since the lesion was quite large and arose from the anterior mucosal wall, we deemed that it was not amenable to resection by conventional laparoscopic approaches. We therefore performed an exploratory laparotomy and exposed the cyst via an anterior gastrotomy. Aspiration revealed 40 mL of thick serosanguinous fluid, suggesting recent hemorrhage into the cyst. The cyst was then resected except for a small area that shared a common wall with the stomach. The remaining cyst was fulgurated, and the gastrotomy and abdomen were closed. Pathologic results for the excised tissue were consistent with gastric duplication cyst and demonstrated gastric antral type mucosa, comprised of muscularis mucosa, submocosa, and fibromuscular tissue. There was no evidence of atypia or premalignant cells.

The cause of gastrointestinal duplication cysts is still debated. Embryologically, they may arise from a persistent diverticulum or aberrant recanalization of the gastric lumen.¹ While large duplication cysts may be diagnosed prenatally or lead to early diagnosis because of symptoms, smaller cysts may remain undetected until adulthood. Various unique presentations including abdominal pain, abdominal mass, bleeding,² and inflammation¹ have been described. The patient's subacute presentation of this congenital anomaly was unusual. We surmise that the recent hemorrhage led to the relatively sudden onset of her symptoms.

Gastric duplication cysts can harbor malignancies such as adenocarcinoma,¹⁰ squamous cell carcinoma,¹¹ or neuroendocrine tumors.¹² While invasion of the serosa of the stomach wall may occasionally signal malignancy,¹⁰ it is usually difficult to predict which cysts will harbor a malignant neoplasm prior to resection. Since malignant neoplasms have been reported even in young children¹³ and the tumors may become metastatic,¹⁰ the recommendation is to perform a resection at the time of diagnosis if the patient does not have significant comorbidities. In summary, the diagnosis of gastric duplication cyst should be considered in the differential diagnosis of gastric outlet obstruction in any age group.

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Correspondence: Tippi C. MacKenzie, MD, University of California, San Francisco, Division of Pediatric Surgery/Fetal Treatment Center, 513 Parnassus Ave, San Francisco, CA 94143-0570 (tippi.mackenzie@ucsfmedctr.org).

Accepted for Publication: August 4, 2009.

Author Contributions:Study concept and design: Zaid, Hirose, and MacKenzie. Acquisition of data: Zaid, Rhee, and MacKenzie. Analysis and interpretation of data: Zaid and MacKenzie. Drafting of the manuscript: Zaid, Rhee, Hirose, and MacKenzie. Critical revision of the manuscript for important intellectual content: Hirose and MacKenzie. Administrative, technical, and material support: Zaid, Rhee, and MacKenzie. Study supervision: Hirose and MacKenzie.

Financial Disclosure: Dr Rhee reports being a member of the Speaker's Bureau for Abbott Nutrition.

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