GASTRIC DUPLICATION CYST

GASTRIC DUPLICATION CYST - A Rare Cause of Gastric Outlet Obstruction

DHARMENDRA SINGH*, SANGRAM SINGH*, SJ KARMARKAR**
*Resident; **Associate Professor and Unit Head, Department of Paediatric Surgery, BJ Wadia Hospital for Children, Parel, Mumbai - 12.

Gastric duplication (GD) is an uncommon malformation of gastrointestinal tract, usually presenting as gastric outlet obstruction. It is usually cystic, attached to the greater curvature of the stomach and has no communication with the stomach. We present a rare case of gastric outlet obstruction secondary to gastric duplication cyst. We have discussed the embryology, clinical features and management of gastric duplication cyst with review of pertinent literature.

INTRODUCTION

Gastrointestinal duplications constitute a rare group of malformations, which vary in site, size, appearance and symptomatology. In 1733 Calder,[1] first reported intestinal duplication. Ladd[2] introduced the term "duplication of the intestinal tract" to encompass a group of congenital anomalies that have three characteristics. Firstly, they have a well developed coat of smooth muscle; secondly, their epithelial lining represents some part of the alimentary tract and thirdly, they are attached to some part of the alimentary tract. Duplications are either cystic or tubular in shape. In 1970, Richard et al[3] presented 9 cases of GD which they saw over 12 years. They identified certain consistent clinical features which have provided the basis for increased recognition of this anomaly. We present a case of gastric duplication that we successfully managed surgically, and the patient is doing well at 2 years of follow up.

CASE REPORT

A second born female patient got admitted in our hospital with complaints of (as given by the parents) non-projectile, gastric (non-bilious) vomiting for the last 5 days. Patient was born of a nonconsanguineous marriage at full term per vaginally. Antenatal history was not significant. There was no family history of pyloric stenosis or other congenital anomalies.

The baby was well nourished, weighing 5.9 kg and had minimal signs of dehydration on admission. There was slight upper abdominal distension. There was no lump palpable nor there were visible loops and peristalsis.

X-ray abdomen showed a normal airfluid level in the stomach and paucity of gases in rest of the abdomen (Fig. 1). USG abdomen revealed a nicely walled off cystic lesion measuring 3.5 x 2.0 x 2.0 cm attached to the greater curvature of the stomach near pylorus.

CT scan abdomen showed a well circumscribed low attenuation fluid collection seen in relation to the greater curvature of the stomach with rim enhancement, suggestive of gastric duplication (Fig. 2). Patient was taken up for surgery and peritoneum was opened by supraumbilical transverse incision. During surgery the lesser sac was opened and an eccentric bulge was seen on the greater curvature in the prepyloric region extending onto the posterior wall. An attempt to develop a plane between the wall of the stomach and the adherent cyst was made but it was not possible. Hence cyst was aspirated and the cyst wall was excised and subjected to biopsy. The epithelial lining of the cyst sharing the common wall with stomach which was left in situ was cauterized.

Histopathological examination revealed gastric mucosa with small pancreatic inclusion and the diagnosis of gastric duplication cyst was made with ectopic pancreatic tissue.

Patient was discharged with normal gastrointestinal function on 10th postoperative day. Patient is now 2 years and 4 months of age and on regular follow up is doing well. Her appetite is good and she now weighs 13 kg.

Fig.1: X-ray abdomen showing air fluid level in the stomach and paucity of gases in rest of the abdomen. Fig.2: CT scan of abdomen showing a well circumscribed low attenuation fluid collection in relation to greater curvature of stomach with rim enhancement.

DISCUSSION

GD is one of the rare causes of gastric outlet obstruction in the neonates. The other causes of gastric outlet obstruction being congenital hypertrophic pyloric stenosis, congenital antral web and in a newborn pyloric atresia which should be included in the differential diagnosis.

Duplications can be found along the entire alimentary tract, the most common site being the ileum. Various theories have been postulated regarding the origin of duplication cysts, like; 1) abortive attempts of twinning;[4] 2) phylogenetic reversion;[5] 3) adhesions between endoderm and neuroectoderm;[6] 4) persistence of embryonic diverticula;[7] 5) recanalisation and fusion of longitudinal folds.[8]

GD usually presents with an upper abdominal mass and vomiting.[3] In our case as the duplication cyst was attached to the posterior wall, mass was not palpable but patient had upper abdominal distension and gastric vomiting which was suggestive of gastric outlet obstruction. A few patients of GD can also present with haematemesis and melaena. Older patients may present with pain in abdomen, nausea and vomiting and weight loss. GD is commonly seen in females.[3]

The diagnosis should be suspected when an infant usually a female comes with features of gastric outlet obstruction. USG and CT scan of abdomen are highly contributive in the diagnosis of GD cyst. We found in literature that GD are managed surgically by simple excision,[3] by dissecting the common wall between the stomach and the duplication cyst, and usually it can be done easily without entering the stomach. But in cases where one does not get a plane of dissection in the common wall one should excise the common wall and suture the gastric defect as leaving behind the common wall (gastric epithelium) would expose the child to inflammatory complications. However, in our case we did marsupialization and cauterization of the epithelial lining instead of excision as we could not get a plane in the common wall. We felt it appropriate because of three reasons. Firstly, excision of common wall in our case would have meant hemigastrectomy (excision of lower half of stomach and pylorus) which would have exposed the infant to associated complications and morbidity of gastroduodenal anastomosis; secondly, it would have increased the anaesthesia and surgical time, and thirdly, it would have increased the hospital stay and the cost of treatment. We have been following up the patient for the last 2 years and patient is growing well, has gained weight and has not experienced any complications so far.

Hence to conclude, we summarize that one should suspect GD, if a female infant presents with features of gastric outlet obstruction. Marsupailization and cauterization of the epithelial lining is a simple safe and effective treatment option wherever it is difficult to find a plane of dissection in the common wall.

REFERENCES

1.Calder J. Medical essays obser 1733; 1 : 205.

2.Ladd WC. Duplication of the alimentary tract. South Medical Journal 1937; 30 : 363.

3.Richard MK, Ronald BL, Robert JI, Jr. JPS 1970; 5 : 3.

4.Edwards H. Congenital diverticula of the intestine with the report of a case exhibiting heterotopia. Brit J Surg 1929; 17 : 7.

5.Hudson HW, Jr. Giant diverticula or reduplications of the intestinal tract. New Eng J Med 1935; 213 : 1123.

6.McLetchie NGB, Purves JK, Saunders RL. Genesis of gastric and certain intestinal diverticula and enterogenous cysts. Surg Gynae Obstet 1954; 99 : 135.

7.Lewis FT, Thyng FW. The regular occurrence of intestinal diverticula in the embryos of pig, rabbit and man. Amer J Anat 1907; 7 : 505.

8.Bremer JL. Diverticula and duplications of the intestinal tract. Arch Path 1944; 38 : 132.

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Fig.1: X-ray abdomen showing air fluid level in the stomach and paucity of gases in rest of the abdomen.	Fig.2: CT scan of abdomen showing a well circumscribed low attenuation fluid collection in relation to greater curvature of stomach with rim enhancement.