Gastric duplication cyst in adult: Challenge for surgeons.

Gastric duplication cysts (GDCs) are uncommon developmental anomalies found primarily in children, being rarely seen in adults. Duplications can occur anywhere in the intestinal tract from the mouth to the anus. Accurate diagnosis of cysts before resection is difficult even using the most advanced imaging techniques. In this report, we present and discuss a case of GDC in a 25-year-old man treated laparoscopically. Patient admitted to our department with complaints of epigastric pain and swelling. Magnetic resonance imaging performed for accurate characterisation showed a 4 cm × 4.5 cm cystic lesion, with heterogeneous signal intensity on T2-weighted images, located in the posterior wall of the stomach. Pre-operative differential diagnosis including gastrointestinal stromal tumour (GIST) was made according to radiological findings. Patient underwent surgery and cyst resected laparoscopically. Histopathological examination suggesting duplication cyst. GDC can easily be mistaken for a GIST, and the clinician as well as radiologist must maintain a high degree of suspicion.

INTRODUCTION

Gastric duplication cysts (GDCs) are uncommon developmental anomalies found primarily in children, being rarely seen in adults. Duplications can occur anywhere in the intestinal tract from the mouth to the anus. Accurate diagnosis of these cysts before resection is difficult even using the most advanced imaging techniques. Differential diagnoses are varied including gastrointestinal stromal tumours (GISTs), neuroendocrine tumours, pancreatic heterotopia, pancreatic pseudocysts, neurogenic tumours and mesenteric cysts. Malignant transformation has been reported very rare in the literature.[12] In this report, we present and discuss a case of GDC in an adult leading challenge for the surgeon, treated laparoscopically.

CASE REPORT

A 25-year-old Caucasian man was admitted to our outpatient clinic with complaints of epigastric pain and swelling. The patient's past medical history and physical examination were non-specific. The findings of routine laboratory tests were all normal. Upper gastrointestinal endoscopy did not reveal any abnormality. Abdominal ultrasonography (USG) which demonstrated a well-defined cystic lesion measuring 4 cm × 5 cm attached to the posterior wall of the greater curvature of the stomach. The patient underwent abdominal computed tomography (CT), which revealed a 40 mm × 46 mm × 46 mm well-marginated cystic lesion suspected to be GIST. Magnetic resonance imaging (MRI), performed for accurate characterisation, showed a 4 cm × 4.5 cm cystic lesion, with heterogeneous signal intensity on T2-weighted images, located in the posterior wall of the stomach [Figure 1a]. Serum levels of carcinoembryonic antigen and carbohydrate antigen 19-9 were both normal. Pre-operative differential diagnosis including GIST was made according to radiological findings. The patient was placed in a supine position. A 10-mm 30° angle camera port was inserted at the umbilicus, one 5-mm port was placed at the right costal margin and done 10-mm were placed at the left costal margin in the midclavicular line for insertion of a harmonic scalpel (Ethicon Endo-Surgery, Cincinnati, OH, USA) instrument. By dividing the gastrocolic ligament, the cyst was visualised from lower margin primarily and after carefully dissected from its dorsal attachments. The gastrosplenic ligament and short gastric vessels were dissected and the greater curvature mobilised. Finally, the whole greater curvature was mobilised, the cyst and the stomach wall was transected using linear endoscopic stapler (Echelon Flex™ 60mm; Ethicon Endo-Surgery, Cincinnati, OH, USA) [Figure 1b]. The post-operative course was uneventful. The patient was discharged on the fifth post-operative day. Histopathological examination demonstrated a benign cystic lesion covered by respiratory epithelium transitioning focally into gastric mucosa with inflammation and microfoci of haemorrhages and intact smooth muscle layer, suggesting duplication cysts [Figure 1c].

Figure 1

(a) T2-weighted magnetic resonance image of the cyst. (b) Laparoscopic view of the cyst. (c) Histopathological view (H and E, × 200)

DISCUSSION

The first report of a GDC was published by Wendel in 1911. Duplications of the gastrointestinal tract are uncommon congenital anomalies and mostly occur in the ileum and followed by the oesophagus, jejunum and colon. Gastric duplications are the least common (3.8%) type of intestinal duplications. Most are non-communicating, single and located along the greater curvature. Approximately, 50% of the patients with GDCs are associated with other congenital anomalies such as oesophageal and duodenal diverticula, duplication cysts elsewhere in the digestive tract, annular and ectopic pancreas or vertebral anomalies such as spina bifida.[3] The aetiology of GDCs is controversial. The mostly noted in hypotheses ranging: The persistence of epithelial outpunching, fusion of embryologic longitudinal folds, endodermal-neurodermal adhesions, sequestration of embryologic tissues during embryonic movements, intestinal ischaemia in early intrauterine life, impaired separation of the notochord from intestinal endoderm, the formation of neurenteric bands with embryonic growth, producing traction diverticula and failure of recanalisation of the bowel lumen following the so-called solid-epithelial phase of the intestinal development, respectively.[4]

The presenting symptoms include epigastric pain, epigastric fullness, vomiting, weight loss, dysphagia, dyspepsia and anaemia. On physical examination, a palpable abdominal mass is most typically found. Occasionally, complications such as gastric outlet obstruction, gastric perforation and carcinoma arising in the cyst can be seen.[5]

Although it is difficult to get a pre-operative diagnosis of GDC, advanced imaging modalities have provided some informative findings. A rate of CT misdiagnosis ranging from 43% to 70% of cases has been reported. MRI does not seem to significantly improve diagnostic accuracy. Endoscopic ultrasonography (EUS) is helpful in identifying the intramural or extramural relation of the gastrointestinal tract. Moreover, EUS-guided fine needle aspiration provides cytological diagnosis. In our case, we could not use EUS since technical failure but other radiological tools such as USG, CT or MRI could not make definitive diagnosis of GDC.[67]

Complete resection of GDC is mandatory with both open and laparoscopic approaches.[8] Advancements in minimally invasive surgery have resulted in application of these techniques even in children, which conveys the known advances of minimally invasive surgery including less wound pain, decreased hospital stay and faster post-operative recovery and less wound-related complications. In our case, we managed a safe and effective resection of GDC laparoscopically. Ineffective approaches including percutaneous or endoscopic aspiration of cystic fluid have been reported but were associated with complications such as fistulaformation and haemorrhage.[78]

GDC can easily be mistaken for a GIST and the clinician as well as radiologist must maintain a high degree of suspicion. Although these lesions are very rare, GDC should be included in the differential diagnosis of gastric wall mass. Laparoscopy must be considered of a safe, possible surgical treatment for GDC.

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Conflicts of interest

There are no conflicts of interest.