Abstract
Intussusception in adults is rare, accounting for 1-2% of intestinal obstruction, often caused by neoplasia. Jejuno-jejunal intussusception is especially uncommon and usually linked to benign lesions. Symptoms are vague, including colicky abdominal pain, vomiting and gastrointestinal bleeding, often delaying diagnosis until exploratory laparotomy. We present two cases of adult jejuno-jejunal intussusception caused by inflammatory fibroid polyps (IFPs), rare benign submucosal tumours mostly found in the stomach and rarely (4.3%) in the jejunum. The first case involved a 17-year old male with abdominal pain, melaena, anaemia and renal dysfunction; surgery revealed a jejunal mass, confirmed as IFP. The second case was a 34-year old female with bowel obstruction from a jejunal mass also identified as IFP. Imaging may mimic malignancy such as gastro-intestinal stromal tumor (GIST) or lymphoma, but histopathology confirms diagnosis. Surgical resection is curative with excellent prognosis.
Keywords
Introduction
Intussusception occurs when a more proximal portion of the bowel (intussusceptum) invaginates into the more distal bowel (intussuscipiens). 1 It is common in children but rare in adults, where it has different causes and symptoms. 2 The latter accounts for 5–12% of all cases and 1–2% of all cases of intestinal obstruction in adults. 3 This may present typically with intermittent bowel obstruction with negative clinical and radiological findings. 4 Symptoms may be vague for a long period and are often diagnosed only during laparotomy for obstructive symptoms. Neoplasia, both benign and malignant, is the leading cause of intestinal intussusception in adults.5,6 Jejunum is an uncommon site where the cause is usually a benign lesion. 7
Case report 1
A 17-year old male presented as an emergency with peri-umbilical pain, vomiting and weight loss for the past three months with significant weight loss. Melena was present over the last month with significant blood loss in the past three days. On examination he was of thin build and anaemic with muscle wasting. He was tachycardiac (120/min), hypotensive (98/60 mm Hg) with normal saturation (98% on air). His abdomen was scaphoid, and a transient peri-umbilical swelling 4 × 4 cm in diameter was evident. There was no guarding.
Laboratory parameters showed a severe anaemia (Hb 62 g/L, a leucocytosis of 27 × 109/L with 92% polymorphs, urea 52.8 mmol/L, creatinine 389 µmol/L, sodium/potassium ratio 144/3.1 mmol/L). Ultrasonography showed telescoping of bowel suggestive of an ileo-ileal intussusception without significant proximal bowel obstruction. Within the lumen of the bowel loop was a homogeneously echogenic soft tissue lesion of 34 ×27 mm. These findings were confirmed by computed tomography scan Fig. 1(a)).
(a) CT (transverse section) showing ‘Target Sign’ and dilated loops; (b) jejuno-jejunal intussusception and (c) a firm mass (IFP) approximately 4 cm in length, the cause of intussusception. CT: computed tomography; IFP: inflammatory fibroid polyp.
After fluid resuscitation and blood transfusion, an exploratory laparotomy was carried out, where a 4 × 3 cm jejunal mass was palpated 70 cm distal to the duodenal flexure (Fig. 1(b) and (c)). This jejunal segment was resected and a stapled jejuno-jejunal anastomosis made. The post-operative period was uneventful.
Histopathology showed that the lesion was arising from the muscularis mucosa and composed of short spindle cells in fascicles with onion skin around blood vessels with no mitosis or necrosis. A significant number of eosinophils were present in the parenchyma. The features were consistent with inflammatory fibroid polyp.
Case report 2
A 34-year old lady presented as an emergency with abdominal distension, vomiting and total intestinal obstruction for two days. Her vital signs were normal, but the abdomen was distended and bowel sounds increased. Radiographs showed multiple bowel-air fluid levels. At emergency laparotomy, an intussusception was found 120 cm from the duodenal flexure and on palpation a solid mass was felt of 4–5 cm in size. The involved jejunal segment was resected and a jejuno-jejunal anastomosis was performed. The post-operative period was uneventful. Histopathology reported an inflammatory fibroblastic polyp.
Discussion
Inflammatory fibroid polyp (IFP) is a benign tumour of the digestive tract, first describe as Vanek's tumour in 1949 as an eosinophilic submucosal granuloma. 8 Its aetiology is unknown. A likely cause is a localised form of gastro-enteritis marked by eosinophilic infiltration, possibly resulting from an uncontrolled inflammatory response to chemical, traumatic or metabolic injury to the mucosa.9,10 The most common location is the stomach in 70% followed by the small bowel 20% and colon 8% but very rare in the duodenum and jejunum. Only 4.3% (12 out of 278) were found in jejunum in a systematic review. 11
The clinical presentation depends on size, position and complications. Most gastric and colonic IFPs are incidentally detected during endoscopy or colonoscopy. Small intestinal IFPs most constantly present with intussusception, though the jejunum is a rare site. 12 They have an average size of 4 cm but giant IFPs up to 20 cm have been reported. 13 Symptoms depend on their site. 14 Small IFPs often go unnoticed on imaging. Ultrasound is useful especially in children, whereas CT is investigation of choice for adults. 15 Under high magnification histopathology, the lesion often shows a whorled or ‘onion-skin’ pattern of cells surrounding blood vessels.16,17
Footnotes
Abbreviations
Authors contributions
LB conceptualised the manuscript. LB and MS reviewed the literature, analyzed the data and made major contributions to the writing of the manuscript. LB, MS and PB performed the clinical examination, surgical treatment and clinical follow up. LB, MS and PB performed the final review and editing of the manuscript. VM provided the HPE diagnosis. All authors have read and approved the final version of the manuscript.
Consent for publication
Written informed consent for publication of the case report and for accompanying images was obtained from the patient. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
Declaration of conflicting interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Ethics approval and consent to participate
Written informed consent for the publication of this case report was obtained from the patient. Approval of the case report by the institutional ethics committee is not required.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
