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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 14  |  Issue : 3  |  Page : 135-137

A rare primary presentation of crohn's disease: Enterocutaneous fistula with abdominal abscess


1 Department of Urology, Dubai Hospital, Dubai, UAE
2 Department of General Surgery, Ministry of Health and Prevention, Kuwait Hospital, Sharjah, UAE
3 Department of Surgery, Tawam Hospital in Affiliation with Johns Hopkins Medicine, Dubai, UAE

Date of Submission21-Dec-2020
Date of Decision22-Mar-2021
Date of Acceptance05-Apr-2021
Date of Web Publication01-Oct-2021

Correspondence Address:
Yazan Ghazi Al Shaikh
Department of Urology, Dubai Hospital, 222 Al Khaleej Road – Deira, Dubai
UAE
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/hmj.hmj_99_20

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  Abstract 


Crohn's disease (CD) is defined as a chronic gastrointestinal tract inflammatory disease which most commonly affects the distal ileum. It might present with a wide range of unspecific symptoms such as diarrhoea, abdominal pain and anorectal pathologies. In this article, we report the case of a 57-year-old man who presented solely with an abdominal abscess associated with low-grade fever. Radiological studies revealed an enterocutaneous fistula and severely inflamed small bowel loops, which were all managed surgically. The patient was stable post-operatively, and final laboratory results confirmed the diagnosis of CD and he was treated accordingly.

Keywords: Abscess, acute abdomen, Crohn's, fistula


How to cite this article:
Al Shaikh YG, Zaki ST, Kawaf Kalla LR. A rare primary presentation of crohn's disease: Enterocutaneous fistula with abdominal abscess. Hamdan Med J 2021;14:135-7

How to cite this URL:
Al Shaikh YG, Zaki ST, Kawaf Kalla LR. A rare primary presentation of crohn's disease: Enterocutaneous fistula with abdominal abscess. Hamdan Med J [serial online] 2021 [cited 2021 Dec 7];14:135-7. Available from: http://www.hamdanjournal.org/text.asp?2021/14/3/135/327431




  Introduction Top


Crohn's disease (CD) also defined as regional enteritis or terminal ileitis is a chronic inflammatory disorder with a relapsing and remitting pattern that might affect any part of the gastrointestinal (GI) tract, although most commonly at the level of the terminal ileum. The clinical presentation as well as the signs and symptoms of CD can be mixed with many other GI-related disorders such as ulcerative colitis and irritable bowel syndrome.[1],[2] Nearly 80% of CD patients present with small intestine involvement, with one-third having ileitis. Moreover, around 50% of the patients might present with ileocolitis with engagement of both the ileum and colon. On the other hand, involvement of the rest of the GI tract such as the oesophagus, stomach or duodenum is not common and is usually linked to other disorders.[3] In this article, we report a case of CD presenting initially as an abdominal wall abscess in the right lower quadrant (RLQ) with enterocutaneous fistula formation.


  Case Report Top


A 57-year-old man presented to the emergency room with complaints of abdominal wall swelling and tenderness in the RLQ. Symptoms started few weeks back with gradual development of pain and low-grade fever. Prior to presentation, he noticed a painful swelling which aggravated mainly on sneezing and movement. The patient denied any nausea, vomiting, change in bowel habits, urinary symptoms or trauma to the area. Past surgical history was significant to an open appendectomy and open resection of bladder mass 1 year back which turned to be benign. Physical examination revealed a warm, tender, immobile 5 cm × 10 cm cystic swelling in the RLQ with mild erythema of the overlying skin. Hernial orifices were intact. Per rectal examination was insignificant. Complete blood count was done and showed leucocytosis (10.12 × 103) and low haemoglobin readings (11.50 g/dL), otherwise laboratories were within normal range.

Abdominal X-ray was done, and findings were unremarkable. After that, computed tomography scan was done with intravenous (IV) and oral contrast. It showed hyperenhancement of the distal ileal loop mucosa and mural stratification with engorged vasa recta. The distal ileal loops appeared oedematous and had thickened walls till the ileocecal valve with 25 cm of luminal narrowing. There was also enhancement of a fistulous tract connected with right-side abdominal wall abscess [Figure 1]a and [Figure 1]b. The patient was then admitted, kept nothing by mouth (NPO) and started on IV broad-spectrum antibiotics and analgesia. He was booked for surgery the next morning.
Figure 1: (a) Transverse view of computed tomography abdomen showing the right abdominal wall abscess along with enterocutaneous fistula formation. Ileal loops are markedly oedematous and inflamed. Intravenous and oral contrasts were already given to the patient. (b) Coronal and sagittal planes of computed tomography abdomen showing more details about the right lower quadrant abscess

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In the operation room, the patient approached laparoscopically at first, but then, it was converted to an open laparotomy as he was found to have multiple peritoneal adhesions. The terminal ileum was markedly enlarged and inflamed as the small bowel loops were entrapped and adherent to the ascending colon and the ileocecal valve. A small rounded mass was also found attached to the anterior abdominal wall, lying deep between muscular planes in the RLQ, at a possible area of enterocutaneous fistula. Incision and drainage were performed, and significant amount of thick pus drained out. Resection of 42 cm of the terminal ileum with end-to-end anastomosis was done in the healthy bowel tissue.

Post-operative period was uneventful, and the patient was improving gradually. Faecal calprotectin level was sent, and it came back as high. The histopathology specimen sent showed partial to completely ulcerated mucosa, intense chronic inflammatory cell infiltration consisting of plasma cells, macrophages and transmural involvement of the bowel wall by lymphoid infiltrates. Granulomas and crypt abscess were seen. No evidence of malignancy was seen. The patient was then referred to a gastroenterologist for further care, and the diagnosis of CD was confirmed.


  Discussion Top


CD as mentioned earlier is a chronic, relapsing and remitting inflammatory disease of the GI tract. Any sections of the GI tract can be affected, typically the terminal ileum like in our case. Most patients present initially with an inflammatory disease, however, complications such as fistulas, strictures or abscesses might develop later in almost half of CD patients, often requiring a surgical intervention. Multiple factors contribute to the aetiology of CD including environmental factors, genetic susceptibility and the nature of intestinal microflora which might affect the mucosal immune response by compromising the epithelial layer function.[4]

The most common initial clinical presentation is a young patient with RLQ associated with change in bowel habits, weight loss and anorexia. If the colon is also involved, rectal bleeding or bloody diarrhoea is commonly present. Extra-intestinal features might be present as well such as arthralgia and oral ulcers.[5] In our case, the atypical presentation for the patient was that he was older than the average age, he did not complain of typical symptoms of CD and only presented due to a complication of the disease. Moreover, the patient denied any family history of the disease. Saying that, CD should always be regarded as a differential diagnosis, especially in the presence of non-specific GI symptoms.

CD workup includes a combination of biopsies review, in addition to clinical, laboratory, radiographic and endoscopic studies. Macroscopically, it is characterised by 'skip lesions' and 'cobblestone' lesions, both of which are unique for CD. Microscopically, granulomas are present in 15%–85% of sampled surgical specimens. However, granulomas can occur also in other conditions like tuberculosis, and therefore, confirming the diagnosis of CD can be a challenge.[6]

Furthermore, faecal calprotectin assesses the disease activity as its concentrations correlate with neutrophil infiltrates in the small bowel and thus represent a very substantial marker of intestinal inflammation with high sensitivity and specificity for inflammatory bowel disease (IBD) diagnosis.[7] Our patient had a high faecal calprotectin level when investigated which raised the suspicion of CD diagnosis in association with the histopathological findings.

Surgical intervention is one of the management options for IBD, however, its role is reserved for patients with advanced unresponsive disease or for those who do not benefit from medical therapy. In addition to that, patients presenting with CD complications like obstructive symptoms might require a surgical resection as anti-inflammatory medications will not be an optimal option at this stage.[4] In the presence of complications such as fistulas and abscesses, surgical resection is needed as well and therefore that was part of the management plan in our case.

It is also important to note that surgical resection does not fully cure CD or its complications, and post-operative recurrence (POR) does occur with relatively high rates. Smoking is the strongest risk factor for POR, increasing it by two folds. Moreover, history of anorectal diseases, intestinal resection and advanced bowel disease (>50 cm) are all recognised risk factors for POR.[8] The most common reoccurrence site after surgical resection is at the newly established anastomosis and might outspread to reach the proximal part of the remaining ileum.[9]

The surgical approach to this patient was done at the beginning in a laparoscopical approach which then was converted to an open laparatomy once the multiple peritoneal adhesions were found which made it very difficult to visualise and resect the needed area. This conversion was done for the optimal outcome and patient safety. An International drainage and a conservative treatment were not considered in this patient as no previous diagnosis was confirmed. Therefore, treating the abdominal wall collection without any surgical resection of the bowel and obtaining a histopathology report was not considered.


  Conclusion Top


CD is not limited to one clinical presentation and may present with unusual signs or symptoms. Awareness of the possible presentations of the disease is essential for reaching the diagnosis correctly and as early as possible to start patients on the optimum management plan, especially the high-risk group which includes elderlies and those with underlying chronic conditions.

Informed consent

The authors obtained an informed consent from the patient. It was stated that only related information will be shared, without mentioning any personal details in our article.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Amarapurkar DN, Patel ND, Rane PS. Diagnosis of Crohn's disease in India where tuberculosis is widely prevalent. World J Gastroenterol 2008;14:741-6.  Back to cited text no. 1
    
2.
Shi C, Montgomery E, Iacobuzio-Donahue CA. Non-neoplastic and inflammatory disorders of the small bowel. Gastrointestinal and Liver Pathology 2012;161-90. https://doi.org/10.1016/b978-1-4377-0925-4.00014-6.  Back to cited text no. 2
    
3.
Mazza M, Cilluffo MG, Cappello M. Clinical presentation of Crohn's disease. Crohn's Disease 2015:7-14. https://doi.org/10.1007/978-3-319-23066-5_2.  Back to cited text no. 3
    
4.
Torres J, Mehandru S, Colombel J, Peyrin-Biroulet L. Crohn's disease. Lancet 2017;389:1741-55.  Back to cited text no. 4
    
5.
Yu H, Liu Y, Wang Y, Peng L, Li A, Zhang Y. Clinical, endoscopic and histological differentiations between Crohn's disease and intestinal tuberculosis. Digestion 2012;85:202-9.  Back to cited text no. 5
    
6.
Geboes K. Histopathology of Crohn's disease and ulcerative colitis. In: Satsangi J, Sutherland LR, editors. Inflammatory Bowel Disease. 4th ed. Edinburgh, London, Melbourne: Churchill Livingstone Elsevier; 2003. p. 255-76.  Back to cited text no. 6
    
7.
Menees SB, Powell C, Kurlander J, Goel A, Chey WD. A meta-analysis of the utility of C-reactive protein, erythrocyte sedimentation rate, fecal calprotectin, and fecal lactoferrin to exclude inflammatory bowel disease in adults with IBS. Am J Gastroenterol 2015;110:444-54.  Back to cited text no. 7
    
8.
Buisson A, Chevaux JB, Allen PB, Bommelaer G, Peyrin-Biroulet L. Review article: The natural history of postoperative Crohn's disease recurrence. Aliment Pharmacol Ther 2012;35:625-33.  Back to cited text no. 8
    
9.
Rutgeerts P, Geboes K, Vantrappen G, Kerremans R, Coenegrachts JL, Coremans G. Natural history of recurrent Crohn's disease at the ileocolonic anastomosis after curative surgery. Gut 1984;25:665-72.  Back to cited text no. 9
    


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