Laparoscopic ‘sleeve’ caecectomy for idiopathic solitary caecal ulcer mimicking appendicitis

Abstract

Idiopathic ulcer of the caecum is a rare condition of unknown aetiology. Its clinical presentation may mimic various pathologies, including appendicitis, inflammatory bowel disease and caecal malignancy. A definitive diagnosis is rarely established preoperatively, and is usually only confirmed histologically following surgical resection. We report a case of a young patient with caecal ulceration presenting with symptoms and signs of appendicitis, in whom laparoscopic anterior ‘sleeve’ caecectomy was performed to excise an inflammatory-looking mass involving the caecum. Histological examination demonstrated a deep mucosal ulcer and subsequent colonoscopy did not reveal any further pathology.

Background

• Idiopathic solitary caecal ulcer is uncommon and its presentation may mimic pathologies more commonly encountered in the acute surgical take, such as appendicitis or inflammatory bowel disease (IBD)

• Laparoscopy is useful in the emergency setting, particularly when the diagnosis is unclear

• Partial caecectomy can easily be performed laparoscopically in cases such as this, thus avoiding the need for laparotomy or more extensive resection

Case presentation

A 21-year-old woman presented to the emergency department with a 24 h history of abdominal pain. The pain was generalised initially, but had localised to the right iliac fossa after several hours. Associated symptoms included anorexia, nausea and several episodes of loose stool. There was no significant medical or family history. Clinical examination revealed tachycardia, and significant right lower quadrant tenderness in the abdomen with signs of localised peritonism.

Investigations

Blood tests showed mildly elevated inflammatory markers with a C reactive protein of 14 mg/L and white cell count of 12.8×10⁹/L. Ultrasonography of the pelvis demonstrated a large amount of free fluid in the pelvic cavity. The appendix could not be visualised, and the uterus as well as both ovaries imaged normally.

Differential diagnosis

The main differential diagnoses in this case included appendicitis, IBD or pelvic inflammatory disease.

Treatment

Diagnostic laparoscopy was performed for presumed appendicitis. This revealed an abnormal cream-coloured mass of approximately 3×3×3 cm on the anterior surface of the caecum. The appendix appeared normal. Dissection of the mass was started; however, as the mass was noted to be adherent to the anterior surface of the caecum (figure 1), we elected to resect it laparoscopically. Partial or ‘sleeve’ caecectomy was performed using an endoscopic linear cutter (figure 2 and video 1). Routine laparoscopic appendicectomy was also carried out.

Figure 1.

Inflammatory mass adherent to anterior caecum.

Figure 2.

Technique of ‘sleeve’ caecectomy using endoscopic linear cutter.

Video 1.

Download video file ^{(18.2MB, mp4)}

DOI: 10.1136/bcr-2015-211058.video01

‘Sleeve’ caecectomy for inflammatory caecal lesion

Outcome and follow-up

Subsequent histological examination of the specimen demonstrated a deep mucosal ulcer within the caecum, with transmural subacute inflammation and fresh fibrosis. There was also serosal inflammation indicating localised peritonitis. The cause of ulceration was not evident on histology; that is, there were no features to suggest inflammatory, infective or neoplastic origin.

The patient's postoperative stay was uneventful and follow-up showed complete recovery, with no further symptoms. Full colonoscopy was subsequently performed, which did not reveal any further ulcers. Random colonic and terminal ileal biopsies were normal.

Discussion

Idiopathic solitary caecal ulcer is an uncommon pathological condition, which was first described by Cruveilhier¹ in 1832. Very little information exists regarding its prevalence. A case series by Cameron² reported documented cases from ages 18 to 80, with an average age of 37 years. A further series by Blundell and Earnest³ found an equal distribution in males and females. Although such lesions may occur in all parts of the colon, more than 50% of solitary colonic ulcers are found in the caecum and ascending colon,⁴ and these are typically located 2 cm from the ileocaecal valve on the anterior, antimesenteric wall.² The most common symptoms are abdominal pain predominantly localised to the right iliac fossa, thus easily leading to a presumed initial diagnosis of acute appendicitis, as in our patient. Other clinical manifestations include haematochezia, diarrhoea, constipation, or an abdominal mass in the right lower quadrant.

The diagnosis of idiopathic caecal ulcer is not easily determined preoperatively, and is usually only confirmed on histology following right hemicolectomy or ‘wedge’ resection. The differential diagnose of solitary caecal ulcer may include bacterial, viral, fungal and parasitic infections, and inflammatory disease such as Crohn's colitis. Malignancy is a common differential, as is caecal diverticulitis or appendicitis. Specific aetiologies of caecal ulceration such as cytomegalovirus infection, particularly in immunosuppressed individuals, have also been reported. In addition, non-steroidal anti-inflammatory drugs and oral contraceptives have been implicated in reports of solitary caecal ulceration.⁵

Imaging techniques such as barium enema may reveal non-specific abnormalities in up to 75% of cases.⁶ These include segmental narrowing, incomplete filling of the affected segment, or mucosal irregularity. However, the ulcer itself is rarely seen on barium enema, and it is frequently impossible to differentiate between benign colonic ulceration and a malignant process.⁷ CT scanning may guide management by demonstrating a distinct mass or perforation, or may point towards alternative pathologies, but is otherwise of little value in suggesting a specific diagnosis. In the experience of Marn et al,⁶ the preoperative CT findings of surgically proven cases of idiopathic caecal ulcer were largely non-specific—these mainly revealed bowel-wall thickening of the caecum and terminal ileum, with evidence of pericolonic fat stranding. Thus the only method for precisely diagnosing these lesions preoperatively is colonoscopy.

In our case, ultrasonography was performed, in which the appendix was not visualised. No comment was made with regard to any mass closely related to the colon; both ovaries and uterus appeared normal. However, the presence of a significant amount of free fluid in the pelvis indicated ongoing pathology in the right iliac fossa, most likely inflammatory in nature and of bowel origin.

Laparoscopy is utilised widely to manage patients with possible appendicitis. In view of the young age of the patient and the appearance of the inflammatory mass, as well as the fact that the rest of the caecum looked normal, we adopted a more conservative approach with partial laparoscopic sleeve caecectomy, rather than a full right hemicolectomy. Also by preserving the ileocaecal valve, we avoided the occasional postoperative persistent diarrhoea that rarely afflicts such patients. It is vital, however, that such partial resections are utilised with care as these can be oncologically inadequate in patients with cancer, and may lead to postoperative leakage in patients with active IBD.

Learning points.

• Idiopathic solitary caecal ulcer is difficult to diagnose preoperatively, but should be considered in the differential diagnosis when encountering patients with right iliac fossa pain, gastrointestinal bleeding or altered bowel habit.

• With stable patients in whom the diagnosis is confirmed at colonoscopy, conservative management may be feasible.

• In patients whose clinical presentation warrants urgent surgery, laparoscopic exploration should be performed.

• Where findings such as in our case are encountered, ‘sleeve’ caecectomy may easily be performed laparoscopically.