Pyloroduodenal Duplication Cysts: Treatment of Eleven Cases

We read with great interest the publication by Lopez-Fernandez et al[1] concerning their large case series of pyloroduodenal duplication cysts (PDDC). The authors describe patterns of clinical presentation, pathomorphology, and guidelines for the surgical management of PDDC. Vomiting and abdominal pain were the most common symptoms, followed by recurrent pancreatitis. We would like to add a case with different aspects of clinical appearance characterized by perforation, peritonitis, and the development of a conglomerate pseudotumor in the right upper abdomen. This normally developed 4-month-old female infant (body weight, 5.6 kg) presented to our department in a seriously reduced condition. She had exhibited a fever of up to 39.5°C for the past 24 hours. Vomiting was not reported, and the infant had regular bowel movements. The clinical investigation revealed tenderness and protrusion of the upper abdomen. The laboratory investigations confirmed systemic inflammation with leukocytosis of 17,000/µL and an elevated C-reactive protein of 16.8 mg/dL. Lipase was not elevated. The ultrasound investigation and magnetic resonance imaging showed an irregular shaped conglomerate of 6 × 6 cm in the right upper abdomen with a cystic and thick-walled core. The diagnosis of a complicated duplication cyst was correctly established by the radiologist. A laparoscopy was urgently performed and confirmed diffuse distributed putrid fluid between the bowel loops and a conglomerate incorporating the gallbladder and liver. We converted to a laparotomy and identified a perforated, noncommunicating, extraduodenal duplication cyst of spherical shape surrounded by a conglomerate ([Fig. 1]). The thick edematous cyst was completely enucleated without entering the duodenal lumen or the pancreaticobiliary ducts. The postoperative recovery was uneventful, and the girl is healthy 3 months after surgery. Histology of the resected specimen revealed small aberrant pancreatic ducts as well as gastric mucosa ([Fig. 2]) and severe acute and chronic inflammation. The wall of the cyst consisted of smooth musculature, but specific acid-producing gastric glands were not found. The perforation might have been the result of digestive erosion or hematogenous infection of the cyst contents. Perforation of a noncommunicating PDDC is an extremely uncommon event. In their extensive meta-analysis, Chen et al[2] described no comparable case. Retrospectively, we found a cystic structure described in an antenatal ultrasound investigation; however, postnatally, no surgery was attempted.

Fig. 1 Clinical appearance of the perforation surrounded by a rim fibrous tissue.

Fig. 2 Gastric glands inside the wall of the duplication (hematoxylin and eosin, magnification ×100).

In conclusion, our case underlines the importance of the final statement from the Madrid group: timely surgical treatment of PDDC is strongly indicated to prevent unexpected and serious complications.

• References

• 1 Lopez-Fernandez S, Hernandez-Martin S, Ramírez M, Ortiz R, Martinez L, Tovar JA. Pyloroduodenal duplication cysts: treatment of 11 cases. Eur J Pediatr Surg 2013; 23 (4) 312-316
• 2 Chen JJ, Lee HC, Yeung CY, Chan WT, Jiang CB, Sheu JC. Meta-analysis: the clinical features of the duodenal duplication cyst. J Pediatr Surg 2010; 45 (8) 1598-1606