Retrograde jejunogastric intussusception presenting as haematemesis in a patient following pancreaticoduodenectomy

Abstract

Intussusception is defined as the invagination of one part of the gastrointestinal tract into another. Jejunogastric intussusception is a rare phenomenon following major upper abdominal surgery, where its aetiology is not well understood. We describe a 68-year-old woman who presented with abdominal pain and haematemesis on the background of a previous pancreaticoduodenectomy (Whipple procedure) for pancreatic cancer. Gastroscopy demonstrated retrograde jejunogastric intussusception, where part of the efferent jejunal limb had prolapsed into the remnant stomach. As a consequence, this intussuscepted segment had become oedematous and ischaemic. The patient subsequently underwent a laparotomy, where the original gastrojejunostomy was resected, which showed the intussuscepted jejunum. The non-viable portion was removed and a Roux-en-Y anastomosis was created. This case highlights the need to ‘think outside the box’ with respect to differential diagnoses when a patient presents with abdominal pain on the background of previous complex abdominal surgery.

Background

Intussusception in adults is a rare phenomenon and most commonly occurs in children.¹ It can be described as the telescoping or invagination of a proximal part of gastrointestinal tract (intussusceptum) into the lumen of a more distal adjacent part (intussuscipiens).¹ In the adult population, the aetiology of intussusception is most common secondary to either benign (polyps, Meckel’s diverticulum, strictures and adhesions) or malignant (lymphoma, adenocarcinoma and metastatic melanoma) conditions that act as a lead point.¹

Regarding jejunogastric intussusception, Shackman described three variants based on anatomical classification: type I involves the afferent limb (antegrade); type II involves the efferent limb (retrograde) and type III involves both limbs.² Type II is the most common form and occurs in 80% of the patients with jejunogastric intussusception.² The pathogenesis of jejunogastric intussusception is not well understood.³ Potential causative factors include jejunal spasm with abnormal motility, long afferent loop, raised intra-abdominal pressure and retrograde peristalsis of the efferent loop.³

Case presentation

A 68-year-old woman presented with a 1-day history of generalised abdominal pain and multiple episodes of small volume haematemesis. She had been lethargic with reduced oral intake over the preceding 2 weeks. She denied any melaena or per rectal bleeding, and had no urinary or infective symptoms. Her surgical history included a classic non-pylorus preserving pancreaticoduodenectomy (Whipple procedure) 3 years ago for T4 N0 M0 Stage III pancreatic adenocarcinoma, for which she had both neoadjuvant FOLFIRINOX and adjuvant gemcitabine chemotherapy. Her medical history included chronic kidney disease, depression, hypertension and secondary pancreatic insufficiency. She lived at home independently, was a current smoker and a non-drinker.

On examination, she was a well-looking woman and her observations were all within normal limits. Abdominal examination revealed generalised tenderness, maximal around the epigastric/umbilical regions. There was no guarding, peritonism, distension or any hernias.

Initially, she was kept nil by mouth, an esomeprazole infusion was commenced and a CT scan of the abdomen/pelvis was arranged.

Investigations

Her blood tests showed a haemoglobin of 97 g/L, white cell count of 17.0×10⁹/L, urea of 10.8 mmol/L, C-reactive protein of 22 mg/L, lactate of 0.4 mmol/L and an international normalised ratio of 1.2.

The CT scan of the abdomen/pelvis revealed a closed loop of jejunum, which had herniated into the gastric lumen. There was significant bowel wall oedema and distension with associated mesenteric vascular congestion suggestive of jejunogastric intussusception (figure 1).

Figure 1.

Coronal (A) and axial (B) CT scan images of the abdomen/pelvis showing intussusception of jejunum into the remnant stomach. The intussuscepted jejunal segment is associated with significant bowel wall oedema and mesenteric vascular congestion.

An urgent gastroscopy demonstrated that approximately 15 cm of the efferent jejunal limb had retrogradely prolapsed into the remnant stomach, with associated oedema and mucosal ischaemia of the intussuscepted segment (figure 2). Although the intussuscepted jejunal limb appeared viable, due to its friable nature, it was deemed unsafe to attempt endoscopic reduction. A nasogastric tube was subsequently inserted under direct vision and the patient returned to the ward for conservative management.

Figure 2.

Gastroscopy images showing (A) the efferent jejunal limb prolapsed into the stomach and a clear lead point with both efferent and afferent limbs identifiable in the distance and (B) the oedematous and ischaemic intussuscepted segment of the efferent jejunal limb.

Treatment

After a 1-day period of conservative management, the patient continued to have episodes of small volume haematemesis with ongoing abdominal pain. Therefore, the decision was made for definitive operative management. A laparotomy was performed and the gastrojejunostomy was resected en bloc, which demonstrated retrograde intussusception of the efferent jejunal limb into the remnant stomach (figure 3). The necrotic jejunal segment was removed and reconstruction was undertaken with a Roux-en-Y anastomosis. Histological analysis of the resection specimen showed extensive ulceration and ischaemia of the small bowel with no evidence of tumour recurrence. The patient recovered well postoperatively, apart from a superficial laparotomy wound infection, and was discharged home on postoperative day 14.

Figure 3.

Intraoperative laparotomy images showing (A) an external view of the retrograde jejunogastric intussusception of the efferent jejunal limb and (B) the oedematous and ischaemic intussuscepted jejunal limb on resection of gastrojejunostomy.

Outcome and follow-up

The patient was reviewed in the outpatient clinic 3 months following her laparotomy for resection of jejunogastric intussusception. Apart from periodic loose bowel motions, she was recovering well. She was maintaining a good diet, was putting on weight and has had no further episodes of abdominal pain or haematemesis.

Discussion

Clinically, there are two forms of jejunogastric intussusception: acute and chronic.⁴ In the acute form, patients often present with epigastric pain, a palpable abdominal mass and vomiting with or without haematemesis, where incarceration or strangulation of the intussuscepted loop can occur, compromising its vascularity.⁴ In the chronic form, patients may experience similar symptoms to those of the acute form but these are usually transient and the intussusception spontaneously self-reduces.⁴ The patient in this case report most likely had an acute form of jejunogastric intussusception, given her symptoms of acute onset of constant abdominal pain and haematemesis.

According to the anatomical classification of jejunogastric intussusception devised by Shackman, our patient was deemed to have type II intussusception, where a segment of the efferent jejunal limb had invaginated into the remnant stomach in a retrograde fashion.² A number of different aetiologies have been identified to act as the lead point in jejunogastric intussusception, including malignancy.¹ Histology demonstrated that no tumour recurrence was present in the resected specimen, which eliminates carcinoma as a cause of the intussusception in this patient. Although the cause of our patient’s intussusception remains unknown, potential mechanisms include adhesions, strictures or retrograde peristalsis.^{1 3}

Jejunogastric intussusception has been reported as a rare complication following gastrojejunostomy, Billroth II gastrectomy and Roux-en-Y anastomosis in less than 0.1% of cases.³ To date, there has only been approximately 200 published cases of jejunogastric intussusception since it was first described by Bozzi in 1914 in a patient with gastrojejunostomy.⁵ To the best of our knowledge, our case is the first to describe a patient with a retrograde intussusception of efferent jejunal limb into the remnant stomach following classic non-pylorus preserving pancreaticoduodenectomy.

An extensive search of the literature has revealed only three similar but not identical case reports of intussusception in patients following pancreatic surgery. The first case described a patient with retrograde intussusception with a history of pancreaticojejunostomy (Puestow procedure) for chronic pancreatitis and a revision of Roux-en-Y anastomosis for small bowel obstruction secondary to intussusception.⁶ Operative findings showed that the revised Roux-en-Y anastomosis was haemorrhagic and oedematous, where the efferent limb had retrogradely intussuscepted into the anastomosis between the afferent limb and the Roux limb.⁶ The second case described a patient with antegrade intussusception with a history of Whipple and Puestow procedures secondary to chronic pancreatitis and pancreas divisum.⁷ It was found that there was intussusception just distal to the most distal Roux-en-Y anastomosis, which had obstructed two Roux-en-Y limbs.⁷ The third case described a patient with retrograde intussusception with a history of Whipple procedure.⁸ Endoscopic and operative findings demonstrated that a long segment of the afferent jejunal limb had intussuscepted into the remnant gastric lumen.⁸ Similar to our case, all three cases required surgical management of the intussusception.

Patient’s perspective.

When my stomach pain began, I felt very tired and bloated. On vomiting, my pain initially got better but when I started to vomit dark liquid resembling blood, I became worried. The next morning, my son took me to hospital, where tests showed that I needed to have a laparotomy. The operation went well and after spending a number of days in the hospital, I was able to go back home. It has now been 6 months since the surgery and I am feeling good. Thank you to all the hospital staff who did a wonderful job in looking after me.

Learning points.

• Jejunogastric intussusception is a rare phenomenon following upper gastrointestinal surgery.

• The pathogenesis of the intussusception may include the presence of a long afferent loop, raised intra-abdominal pressure or retrograde peristalsis of the efferent loop.

• Prompt initial resuscitation and investigation of abdominal pain and haematemesis with imaging and gastroscopy is essential for the accurate diagnosis and management of jejunogastric intussusception.

• Surgical management of intussusception should not be delayed due to the potential risk of haemorrhage, ischaemia, bowel obstruction and subsequent poor outcomes.

• It is always important to consider alternative differential diagnoses and ‘think outside the box’ especially in patients who present with abdominal pain and a history of complex abdominal surgery.