Safety and Efficacy of Endoscopic Therapy for Nonmalignant Duodenal Duplication Cysts: Case Report and Comprehensive Review of 28 Cases Reported in the Literature

Abstract

Analyze efficacy, safety of endoscopic therapy for duodenal duplication cysts (DDC) by comprehensively reviewing case reports.Tandem, independent, systematic, computerized, literature searches were performed via PubMed using medical subject headings or Keywords "cyst" and "duodenal" and "duplication"; or "cyst", and "endoscopy" or "endoscopic", and "therapy" or "decompression"; with reconciliation of generated references by two experts. Case report followed CARE guidelines.Literature review revealed 28 cases (mean = 1.3 ± 1.2 cases/report). Endoscopic therapy is increasingly reported recently (1984-1999: 3 cases, 2000-2015: 25 cases, P = 0.003, OR = 8.33, 95%-CI: 1.77-44.5). Fourteen (54%) of 26 patients were men (unknown-sex = 2). Mean age = 32.2 ± 18.3 years old. Procedure indications: acute pancreatitis-16, abdominal pain-8, jaundice-2, gastrointestinal (GI) obstruction-1, asymptomatic cyst-1. Mean maximal DDC dimension = 3.20 ± 1.53 cm (range, 1-6.5 cm). Endoscopic techniques included cyst puncture via needle knife papillotomy (NKP)/papillotome-18, snare resection of cyst-7, cystotome-2, and cyst needle aspiration/ligation-1. Endoscopic therapy was successful in all cases. Among 24 initially symptomatic patients, all remained asymptomatic post-therapy without relapses (mean follow-up = 36.5 ± 48.6 months, 3 others reported asymptomatic at follow-up of unknown duration; 1 initially asymptomatic patient remained asymptomatic 3 years post-therapy). Two complications occurred: mild intraprocedural duodenal bleeding related to NKP and treated locally endoscopically.A patient is reported who presented with vomiting, 15-kg-weight-loss, and profound dehydration for 1 month from extrinsic compression of duodenum by 14 × 6 cm DDC, underwent successful endosonographic cyst decompression with large fenestration of cyst and endoscopic aspiration of 1 L of fluid from cyst with rapid relief of symptoms. At endoscopy the DDC was intubated and visualized and random endoscopic mucosal biopsies were obtained to help exclude malignant or dysplastic DDC.Study limitations include retrospective literature review, potential reporting bias, limited patient number, variable follow-up.In conclusion, endoscopic therapy for DDC was efficacious in all 29 reported patients including current case, including patients presenting acutely with acute pancreatitis, or GI obstruction. Complications were rare and minor, suggesting that endoscopic therapy may be a useful alternative to surgery for nonmalignant DDC when performed by expert endoscopists.

FIGURE 1

A, Upper gastrointestinal series with small bowel follow-through in a 40-year-old man presenting with severe nausea and vomiting post cibum associated with 15 kg weight loss during the prior month reveals a smoothly contoured deformity along the third portion of the duodenum caused by a central mass displacing and compressing this part of the duodenum. The duodenum is mostly but not completely obstructed by the central mass. B, Abdominal magnetic resonance imaging (MRI) without IV gadolinium contrast reveals an 8 × 13 cm intramural cystic mass arising along wall of the descending and transverse duodenum that demonstrated T1 and T2 hyperintense signals, and an irregularly thickened posterior cyst wall, findings consistent with a duodenal duplication cyst containing complex fluid, such as hemorrhagic or proteinaceous material.

FIGURE 2

Progressive stages of endosonographic therapy to decompress a highly symptomatic and very long duodenal duplication cyst. A, Endoscopic ultrasound (EUS) revealed an oval, intraluminal (subepithelial), anechoic lesion, that endosonographically originated from within the submucosa (layer 3), consistent with duodenal duplication cyst. The cyst arose from muscularis propria of descending duodenum just distal to the papilla, extended deeply into the third portion of duodenum, and contained significant debris within it. B, The duodenal cyst was punctured using a 19 gauge needle; thick, dark fluid was aspirated; a 0.035 in. Jagwire was advanced via the needle into the cyst; the needle was withdrawn; a needle-knife papillotome was fed over the Jagwire; and the cyst wall was incised using the papillotome. C, The papillotome was exchanged with a pyloric balloon dilator, and the aperture was serially dilated up to 18 mm using increasingly larger balloons. D, EGD reveals a wide aperture after progressive balloon dilatation. E, The echoendoscope was withdrawn and a pediatric colonoscope was intubated and advanced into the cyst, and 800 cc of dark, brown fluid was aspirated via the colonoscope This endoscopic photograph shows the mucosa within the cyst has no evident lesions after cyst aspiration.