Acute massive gastric dilatation: a rare, forgotten complication of fundoplication

Abstract

An 8-year-old boy with a history of multiple neonatal laparotomies, including congenital diaphragmatic hernia repair and an open fundoplication, presented acutely with severe abdominal pain, distension, vomiting and shock. A large abnormal opacity in the left upper quadrant was visible on a plain abdominal radiograph. The patient was taken to the theatre for emergency laparotomy and was found to have a massively distended stomach, the fundus and body of which were necrotic. A subtotal gastrectomy was performed, sparing the viable tissue. The patient went on to make a full recovery. Acute massive gastric dilatation (AMGD) is a rare condition characterised by severe gastric distension. Gastric ischaemia results when intragastric pressure exceeds venous pressure, obstructing venous outflow. It is important to recognise AMGD as a severe complication of fundoplication due to closed-loop gastric obstruction. It should prompt consideration of an early laparotomy in cases where the diagnosis is suspected.

Background

Fundoplication is a common antireflux procedure performed in both paediatric and adult populations. In this case report, we highlight acute massive gastric dilatation (AMGD) as a rare potential complication of fundoplication due to closed-loop gastric obstruction with a high risk of mortality. AMGD should be considered in the differential diagnosis in all patients who underwent fundoplication that present acutely.

Case presentation

An 8-year-old boy with a background of multiple neonatal laparotomies presented to the emergency department with several hours of severe colicky abdominal pain, distension and vomiting. On examination, the patient was shocked, with a grossly distended, peritonitic abdomen. A nasogastric tube was passed which failed to drain significant amounts of fluid. Due to the severity of the presentation, the patient was taken to the theatre for emergency laparotomy after aggressive fluid resuscitation.

Background and context

The patient was born at term following antenatal diagnosis of a congenital diaphragmatic hernia (CDH). On day 3 of life, he was taken to the theatre for the primary sutured repair of a large left-sided posterolateral defect transmitting the spleen, stomach, small bowel and colon. His initial postoperative course was complicated by recurrent left lung collapse and prolonged total parenteral nutrition dependence.

An upper gastrointestinal contrast study at 3 weeks of age demonstrated reflux through a significant hiatus hernia and gastric outlet obstruction. He was returned to the theatre for duodenal adhesiolysis in an attempt to correct the latter. A third laparotomy became necessary at 5 weeks due to the recurrence of gastric outlet obstruction. Further adhesiolysis and a duodenoduodenostomy were performed. In the same procedure, the large hiatal defect was closed with sutures, followed by a 270° fundoplication and gastrostomy, then a Ladd’s procedure was performed for a significantly narrowed mesentery. Recovery was uncomplicated and he was eventually established on gastrostomy and oral feeds.

He was discharged from the hospital at 8 weeks of age. He progressed well during follow-up and by 6 months was on complete oral feeds with the gastrostomy removed. He was living a normal life, symptom free, until his acute presentation.

TREATMENT

At laparotomy, the patient was found to have dense adhesions and a massively distended stomach, the fundus and body of which were necrotic. There was a notable lack of a recognisable left gastric artery. Sleeve gastrectomy was performed, removing the necrotic tissue and sparing the viable gastro-oesophageal junction, lesser curve and antrum, which were closed over a button gastrostomy. A feeding jejunostomy was also placed.

Blood gas revealed a lactic acidosis (8 mmol/L). A plain abdominal X-ray was taken which showed generalised small bowel dilatation and a large abnormal opacity in the left upper quadrant (figure 1).

Figure 1.

Plain abdominal radiograph. Stomach visible as large opacity in the left upper quadrant.

Outcome and follow-up

The recovery was complicated by a minor haemorrhage from the gastric suture line; however, this did not require intervention. Jejunostomy feeds were established quickly, with gradually increasing amounts of oral diet, and he was discharged 22 days after the operation. Weaning of jejunostomy feeds took place successfully during follow-up, allowing removal of both the gastrostomy and jejunostomy. The patient is currently doing well, maintaining weight on a normal diet.

Discussion

AMGD is a rare condition characterised by severe gastric distension, leading to the stomach occupying almost the entire space of the abdominal cavity. Gastric ischaemia results when intragastric pressure exceeds venous pressure, obstructing venous outflow.^{1 2} It has a high mortality rate due to its complications: gastric necrosis, emphysema and perforation, the latter is thought to have a mortality rate of 80%–100%. In more than 90% of cases, patients present with vomiting; other symptoms include abdominal pain and progressive abdominal distension. Plain abdominal radiography reveals a distended stomach with an air–fluid level. Hepatic portal venous gas, as a result of gastric emphysema may also be visible on radiographs, CT or, while potentially inappropriate in the acute setting, ultrasound scanning.³ Initial management before surgical intervention includes intravenous fluids and insertion of a nasogastric tube to aid decompression of the stomach.⁴

Although the exact aetiology is unknown, AMGD has been observed in association with several different pathological entities. It is well described in binge eating disorders where the stomach has become atonic and atrophied during periods of starvation. Massive distension then occurs on ingestion of a large meal.^5–7 In other instances, AMGD has been the sequelae of small bowel obstruction, electrolyte disturbances, diaphragmatic hernia, spinal pathology and trauma.⁸ In several cases, it has been caused by a closed-loop obstruction of the stomach with a mix of iatrogenic or pathological causes, for example, proximal obstruction by an oesophageal adenocarcinoma or fundoplication and distal obstruction caused by adhesions, tumours or volvulus around a feeding jejunostomy.^{3 4 9}

Although fundoplication is a well-documented contributing factor in adults, there have been no recent reports of AMGD in children as a result of this surgery. Idowu et al reported a fatal case secondary to jejunal volvulus in a child with a fundoplication.¹⁰ Bass et al described AMGD and gastric necrosis in a neurologically impaired 11-year-old boy with a fundoplication, who was thought to have had an acute-on-chronic event.¹¹ The most recently reported case, by Sowande and Bianchi in 1999, of a 21-month-old patient with a background of CDH repair and fundoplication is similar to our own.¹² In both Sowande and Bass’s cases, there was total gastric necrosis, and while both patients survived, they required gastric replacement surgery.

In the case of our patient, we believe the proximal fundoplication wrap and distal adhesions from previous laparotomies appeared to have precipitated a closed-loop gastric obstruction and AMGD. The ischaemia is likely to have been exacerbated by a pre-existing lack of the left gastric and right gastroepiploic arteries, possibly caused by poor development secondary to herniation of the stomach, or damage while mobilising the stomach during the neonatal CDH repair and fundoplication. As an under-recognised complication, with no reports of AMGD in children with fundoplication for two decades, our case serves as a reminder of the importance of early recognition and surgery to avoid the devastating sequelae of this condition.

Learning points.

• Acute massive gastric dilatation (AMGD) is a rare, forgotten but serious complication of fundoplication.

• AMGD should be considered in any patient with a previous fundoplication presenting with symptoms of acute obstruction as a tight fundoplication with distal bowel obstruction can precipitate closed-loop gastric obstruction.

• Early laparotomy should be considered in patients with suspected AMGD.