Vol 44 # 2 June 2012 - Kma.org.kw

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143 KUWAIT MEDICAL JOURNAL June 2012 Case Report Incomplete Small Bowel Obstruction Caused by Idiopathic Diaphragm Disease of the Proximal Ileum Stewart Gibson, Arin K Saha, Ian F Hutchinson Department of Surgery, Airedale General Hospital, Steeton, United Kingdom Kuwait Medical Journal 2012; 44 (2): 143 - 145 ABSTRACT Diaphragm disease is a rare pathology and describes the phenomenon of circumferential, concentric mucosal lesions of the bowel which can cause progressive narrowing of the bowel lumen and bowel obstruction. The etiology in significant majority of cases is previous or concurrent ingestion of nonsteroidal anti-inflammatory drugs (NSAID). A 55-year-old Caucasian man with no history of prior ingestion of NSAID presented with a three week history of progressive abdominal pain, distension and subsequent absolute constipation. He failed a trial of conservative management and subsequently underwent a laparotomy and small bowel resection for small bowel obstruction. Histopathological analysis revealed diaphragm disease of the small bowel. This case emphasizes the complexities in the management of patients with incomplete small bowel obstruction and highlights the possibility of diaphragm disease of the bowel in the absence of known risk factors. KEY WORDS: gastroenterology, general surgery, diaphragm disease INTRODUCTION Diaphragm disease (DD) is a rare pathology of the gastrointestinal tract, most commonly affecting the small bowel. It is characterized by concentric mucosal lesions (‘diaphragms’) within the bowel which reduce the diameter of the bowel lumen [1] . Although the pathogenesis of the condition is still unclear, nonsteroidal anti-inflammatory drug (NSAID) use is considered as the most common predisposing factor [1] . This case report describes a patient with no known predisposing risk factors who developed diaphragm disease of the small bowel. CASE PRESENTATION A 55-year-old Caucasian man presented to the Emergency Department (ED) of a district general hospital with a three-week history of colicky, central abdominal pain. He had not passed flatus for 48 hours. He had vomited once, two days prior to admission and reported a reduced appetite and significant weight loss over the course of his illness. His past medical history included chronic leg pain since an accident 35 years ago and he required regular co-codamol (30/500 mg, QDS). He also admitted to intravenous drug abuse many years earlier. He reported no previous or recent use of NSAID analgesia and no history of inflammatory bowel disease. Physical examination revealed tenderness in the peri-umbilical region and soft abdominal distension. He had no clinical signs of peritonism and his bowel sounds were audible, though high-pitched. He had no palpable herniae and no palpable masses or organomegaly. Hematological investigations revealed a microcytic anemia (Hemoglobin = 9.0 g/dl, Mean Cell Volume = 63 fl). Plain abdominal radiographs revealed small bowel obstruction (Fig. 1). Urgent computed tomography (CT) imaging was performed which confirmed dilated, fluid filled small bowel loops but also gas and feces in the large bowel with no transition point of true obstruction in the small bowel. Initial management was conservative in nature with nasogastric decompression of the stomach, careful fluid balance and intravenous fluid infusion. His condition worsened and he vomited one liter of feculent fluid four days after admission. He proceeded to emergency laparotomy where the small bowel was dilated proximal to a single, well-defined transition point in the proximal ileum. There was no evidence of inflammatory bowel disease or carcinoma and no extrinsic compression. An intra-luminal stricture Address correspondence to: Arin K Saha, MRCS (Eng), Specialist Registrar, Dept. Of General Surgery, Airedale General Hospital, Skipton Road, Steeton, BD20 6TD United Kingdom. Tel: +44 07799 40 20 10, E-mail: arinsaha@yahoo.com
June 2012 KUWAIT MEDICAL JOURNAL 144 Fig. 2: Histopathological analysis of intra-luminal diaphragm within the small bowel consisting of prominent hyperplastic folds with intervening stromal cells Fig. 1: Plain supine abdominal radiograph which demonstrates dilated small bowel consistent with small bowel obstruction was found at the point of transition with a patent, but very narrow lumen. Similar constrictions were noted throughout the length of the small bowel but the patency of the lumen was not compromised at these areas. A wedge resection of the affected area of small bowel was performed and stapled side-to-side anastomosis using the TLC 75 (Ethicon, UK) device was fashioned. He made a good post-operative recovery and was discharged home four days after surgery. Histopathological examination of the resected specimen revealed the presence of an intra-luminal diaphragm within the small bowel consisting of prominent hyperplastic folds with intervening stromal cells, consistent with diaphragm disease of the small bowel (Fig. 2). The diaphragm consisted of significant fibrosis, which was confined to the submucosal layer with minimal mucosal ulceration. This was confirmed on both hematoxylin and eosin staining and Van Gieson’s staining. There was a slightly greater density of neutrophil polymorphs and lymphocytes at the areas of mucosal ulceration. There was no evidence of any other pathology; in particular, there was no evidence of Crohn’s disease, malignancy or other inflammatory bowel disease and specific immune-histochemistry stains were not performed. DISCUSSION DD was first described in 1988 [1] as circumferential mucosal lesions forming “diaphragms” within the small bowel. The small bowel is most commonly affected although similar lesions have been reported in the colon [2] . Histologically, diaphragms are similar to the normal plicae circulares in the small bowel but with submucosal fibrosis, and occasional mucosal ulceration and villous atrophy [1] . There is a clear association between DD and long-term NSAID use and NSAIDs are known to affect enterocytes directly by increasing intestinal permeability [3] , which might allow bacterial invasion of the bowel wall with secondary inflammation. DD in the absence of NSAID use is very rare although unknown NSAID ingestion was possible in these cases [2,4] . In this case, given the long term management of chronic leg pain, formal documentation of analgesics prescribed both in the community and hospital settings was available and we are confident that there was no prior NSAID use. We are unsure as to the etiology of diaphragm disease in this case and, on histopathological analysis, there was no chronic inflammation or ulceration, suggesting that systemic factors may have a role. Radiological investigations, as in this case, can be misleading and can often overlook intra-luminal diaphragms. The diagnosis is usually confirmed on histopathological analysis although can be suspected from other imaging techniques such as capsule endoscopy, small bowel enteroscopy, small bowel contrast studies or enteroclysis. The tendency for objects to be retained within diaphragm compartments themselves may also be useful for diagnosis [5] . Progressive obstruction caused by diaphragm disease requires surgical resection of the affected bowel and strictuloplasty-type operations are of no benefit given the circumferential nature of the disease. There is no current guidance on the management of other diaphragms found at surgery. In this case, resection was not possible given the length of bowel that was affected and as such an intra-operative decision to leave bowel that had a patent lumen was taken and
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Vol 44 # 2 June 2012 - Kma.org.kw

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