Laparoscopic excision of intra-abdominal oesophageal duplication cyst in a child.

Duplication cysts are congenital cystic malformation of the alimentary tract consisting of a duplication of the segment to which it is adjacent. It can occur anywhere from mouth to anus. Oesophageal duplication cysts comprise 4% of the same. Of these, total intra-abdominal oesophageal duplication cysts are extremely rare. On review of literature, only 3 case reports of total intra-abdominal oesophageal duplication managed laparoscopically are found. All these cases were adults. We report the first paediatric case of intra-abdominal oesophageal duplication cyst excised laparoscopically.

INTRODUCTION

Duplication cysts of GIT are congenital cystic malformation of the alimentary tract, consisting of a duplication of the segment to which it is adjacent, occurring anywhere from the mouth to the anus but most frequently affecting the ileum.[1]

The most common duplication cysts arise from the small intestine (44%) followed by large intestine (15%). The oesophageal duplication cysts comprise around 4% of duplication cysts of GIT. They can be 1) cervical, 2) thoracic and thoraco-abdominal, and 3) total intra-abdominal oesophageal duplication cysts. [Table 1]

Table 1

GI tract duplications[2]

Most common of oesophageal duplication cysts are thoracic and thoraco-abdominal. Only intra-abdominal oesophageal duplication cysts are very rare. Excision is the treatment of choice either by open or by laparoscopic surgery.

CASE REPORT

A 13-year-old girl presented with c/o pain abdomen since 2 days. H/o vomiting 3 episodes and high degree fever. No previous significant history. O/E her vitals were stable. P/A there was tenderness in epigastric region, rest of the abdomen was soft. There was no guarding/rigidity. Bowel sounds and other systems were normal.

Her blood investigations were normal except for slight leucocytosis. Ultrasonography of abdomen showed a cystic lesion in the upper abdomen adjacent to left lobe of liver. Computerized tomography scan of the abdomen and magnetic resonance imaging of abdomen narrowed the differential diagnosis to a cystic lesion at the gastro-oesophageal junction adjacent to left lobe of liver measuring 4 × 5 cm in size. Clinical diagnosis of foregut duplication cyst was made.

Diagnostic laparoscopy and excision of the cyst was planned. A 10 mm umbilical port for camera and two 5 mm working ports on either side with an additional port for liver retraction were used. [Figure 1]

Figure 1

Port sites marked with cyst position

A cystic lesion at gastro-oesophageal junction below the left lobe of liver was identified. The cyst was adherent to oesophagus [Figure 2]. With help of harmonic scalpel, cyst was dissected from surrounding structures. Subtotal excision of cyst wall was done except the part that was adherent to oesophagus. Mucosal layer of the cyst wall adherent to the oesophagus was excised [Figure 3]. Intactness of oesophageal mucosa was confirmed by injecting air through nasogastric tube positioned at lower end of oesophagus. Drain was inserted from right sub costal port site, which was removed after 48 hours. Child had uneventful post operative recovery and discharged from hospital on 6th postoperative day. Histopathology confirmed the presence of muscle layer and lined by gastric mucosa.

Figure 2

Intraoperative view of cyst

Figure 3

Excised cyst

DISCUSSION

Gastrointestinal duplication cysts are seen in 1 of every 4,500 autopsies. Only 4% of these are oesophageal duplication cysts. The duplication cyst of oesophagus can be in the form of a separate tube or a spherical cyst either in continuity or in close proximity of the oesophagus.[3] Total 90% of them do not communicate with the lumen; 80% of such cases are symptomatic in childhood. Acute symptoms are due to perforation, haemorrhage or secondary infection. Presence of aberrant gastric mucosa is the cause for onset of complications. Sudden onset of pain and vomiting are the usual presenting symptoms.[4]

Ultrasonography is the basic diagnostic test; however, CT scan is more accurate especially for intrathoracic cysts. The other modalities of diagnosis mainly include endoscopy, trans-oesophageal ultrasonography and MRI.

Excision is indicated even if the cyst is accidentally detected keeping in view the high incidence of complications. Thoracic oesophageal duplication cysts are excised by thoracotomy or VATS. The intra-abdominal oesophageal duplication cysts are excised by laparotomy or laparoscopically. Postoperative complications include oesophageal diverticulum formation and recurrence if complete excision is not done.[5]

The histopathology confirmation of oesophageal duplication cyst is by Palmer′s criteria: 1) attachment to oesophageal wall, 2) lining of GI mucosa, 3) presence of muscle layer.[3]

Oesophageal duplication cysts are more commonly seen in the thorax. Intra-abdominal oesophageal duplication cysts are very rare. Review of literature till date indicates that only 3 cases are reported of laparoscopic excision of intra-abdominal oesophageal duplication cysts.[6] All the 3 cases were in adults. Probably this is the first paediatric case of total intra-abdominal oesophageal duplication cyst that was excised laparoscopically.

CONCLUSION

Intra-abdominal oesophageal duplication cysts are very rare. Acute symptoms are due to perforation, haemorrhage or secondary infection. Laparoscopic excision is possible even in paediatric age group.