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11/166. Diagnostic difficulties in neuronal intestinal dysplasia and segmental colitis.

    An 11-month-old girl with a prolonged history of bloody, mucoid diarrhea is presented. Although the initial diagnosis given by the rectosigmoid biopsy obtained during laparotomy was neuronal intestinal dysplasia, accompanying findings including mixed inflammatory cell infiltration of the mucosa and submucosa with mucosal ulcerations suggested nonspecific colitis. The subsequent biopsy specimen that was obtained after performing colostomy and treating with broad-spectrum antibiotics and rectal irrigations showed improvement in the structure of ganglion cells and submucous and myenteric plexuses. Although the mucosal ulcerations and inflammatory reaction improved, the colonic stricture persisted, so the Duhamel procedure was performed, and the patient had an uneventful outcome. It is claimed that inflammatory disease of the rectosigmoid colon of unknown etiology and neuronal intestinal dysplasia have occurred together in the current case or that one disease might cause the other in time.
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12/166. diarrhea as a side effect of mesalamine treatment for inflammatory bowel disease.

    tablets of coated 5-aminosalicylate (5-ASA) called mesalazines are among the most widely prescribed preparations for the treatment of colitis, including ulcerative, Crohn's disease (CD), collagenous and lymphocytic colitis, and-to a lesser extent-CD of the small bowel. Mesalamines have, to a large extent, replaced the parent drug sulfasalazine because they produce fewer side effects. Although mesalamines have been known from earlier studies to produce occasional diarrhea, the true incidence of this effect is not known and is not always recognized. We are presenting the cases of five patients in whom oral mesalamines produced severe and persistent diarrhea-made worse by increasing doses of the drug-and intensified the colitis in two. Recognition of the problem is by symptom analysis after careful history taking. Changing the therapeutic regimen or discontinuing the medication is usually required to reverse this potentially debilitating and occasionally life-threatening side effect.
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13/166. Laboratory findings associated with thrombophilia are not more common in inflammatory bowel disease.

    Thromboembolic disease (TED) has been recognized as a complication of inflammatory bowel disease (IBD) since the 1930s (Bargen & Barker 1936). The relative contributions of inherited or acquired thrombophilia and the inflammatory response to the mechanism of this tendency is unclear. Thrombotic events are more common in active disease although significant numbers also occur spontaneously, when the disease is in clinical remission (Talbot et al. 1986; Jackson et al. 1997). Studies looking at the prevalence of specific thrombophilic states such as antithrombin iii deficiency (Jackson et al. 1997; Lake, Stauffer & Stuart 1978; Cianco et al. 1996; Ghosh et al. 1983), factor v Leiden mutation (APC Resistance) (Jackson et al. 1997; Probert et al. 1997; Ardizzone et al. 1998; Liebman et al. 1998), anticardiolipin antibodies (Ciancio et al. 1996), protein c (Wyshock, Caldwell & Crowley 1988; Korsten & Reis 1992) and protein s deficiencies (Jorens et al. 1990; Aadland et al. 1992) in IBD have been contradictory or equivocal. We had previously found that IBD patients with a history of TED are not more likely to have a laboratory thrombophilic abnormality than those with uncomplicated disease. We also demonstrated that the prevalence of heterogenous laboratory thrombophilic abnormalities (usually minor) in all IBD patients may be as high as 60%, much higher than the recognized prevalence of TED (Lim, Jones & Gould 1996). We wondered how this would compare with the healthy non-IBD population. We have therefore explored the prevalence of such thrombophilic abnormalities in a group of IBD patients who had no history of TED and compared them with healthy age and sex matched controls.
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14/166. Eosinophilic pleural effusion due to mesalamine. Report of a rare occurrence.

    mesalamine-induced lung toxicity has often been described. We report on a case of a patient who underwent mesalamine treatment, though in the absence of established criteria required for diagnosing Crohn's disease (CD) or ulcerative colitis (UC). He developed an adverse respiratory reaction to the drug, thus definitely proving its lung damaging capacity. The clinical presentation included eosinophilic pleural effusion, a feature never previously described in association with mesalamine intake.
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15/166. An unusual white blood cell scan in a child with inflammatory bowel disease: a case report.

    technetium-99m-labeled leukocyte (WBC) imaging is a valuable screening method for inflammatory bowel disease, especially in children, because of its high rate of sensitivity, low cost, and ease of preparation. A 14-year-old girl is described who had juvenile arthritis and iritis complicated by inflammatory bowel disease. She was examined for recurrent abdominal pain. A Tc-99m stannous colloid WBC scan was performed, and tracer accumulation was seen in the small bowel in the region of the distal ileum on the initial 1-hour image. Delayed imaging at 3 hours also revealed tracer accumulation in the cecum and ascending colon, which was not seen on the early image. A biopsy of the colon during endoscopy showed no evidence of active inflammation in the colon. The small bowel was not seen. Computed tomography revealed changes suggestive of inflammatory bowel disease in the distal ileum. The appearance on the WBC study was most likely a result of inflammatory bowel disease involving the distal ileum, with transit of luminal activity into the large bowel.
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16/166. bartonella henselae and inflammatory bowel disease.

    Sustained fever and increased thickness of the distal ileum on ultrasound suggested Crohn's disease in an adolescent boy. bartonella henselae infection was diagnosed by specific serology and the patient recovered. ileitis could be related to B. henselae infection.
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17/166. Filiform polyposis of the colon in chronic inflammatory bowel disease (so-called giant inflammatory polyps).

    On the basis of 3 of our own cases, we describe unusually intense forms of filiform polyposis and local giant polyposis as a consequence of chronic inflammatory bowel disease. The patients are: A 52-year-old woman who for 7 years has been known to have Crohn's disease (CD); a 55-year-old man who for 14 years has been known to have chronic inflammatory bowel disease, which was first thought to have been ulcerative colitis, but, as a result of the findings on the subtotal colectomy specimen, had to be classified as Crohn's disease or colitis indeterminate; and a 53-year-old woman known to have had ulcerative colitis for 37 years. From the literature on the subject, we drew up a chronological list of a total of 43 cases with similar or completely identical findings. The clinical significance of the findings in their particularly massive intensity results from their necessary differentiation--in the context of differential diagnosis--from a malignant tumor, in particular from a carcinoma in association with chronic inflammatory bowel disease, or from a villous adenoma. The indication of a need to operate results from the impossibility of being able definitely to rule out a malignant degeneration by means of clinical methods. Also, experience shows that with massive findings of the kind described a spontaneous disappearance cannot be expected. Finally, too, the clinical symptoms and the patients subjective complaints necessitate balanced surgical treatment, taking into consideration the site and the extent of the lesion.
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ranking = 4.5
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18/166. Intravenous cyclosporine in refractory pyoderma gangrenosum complicating inflammatory bowel disease.

    BACKGROUND: pyoderma gangrenosum complicates inflammatory bowel disease in 2-3% of patients and often fails to respond to antibiotics, steroids, surgical debridement or even colectomy. methods: We performed a retrospective chart analysis of 11 consecutive steroid-refractory pyoderma patients (5 ulcerative colitis, 6 Crohn's disease) referred to our practice and then treated with intravenous cyclosporine. pyoderma gangrenosum was present on the extremities in 10 patients, the face in 2, and stomas in 21. At initiation of intravenous cyclosporine, bowel activity was moderate in 3 patients, mild in 4, and inactive in 4. All patients received intravenous cyclosporine at a dose of 4 mg/kg/d for 7-22 days. They were discharged on oral cyclosporine at a dose of 4-7 mg/kg/d. RESULTS: All 11 patients had closure of their pyoderma with a mean time to response of 4.5 days and a mean time to closure of 1.4 months. All seven patients with bowel activity went into remission. Nine patients were able to discontinue steroids, and nine were maintained on 6-mercaptopurine or azathioprine. One patient who could not tolerate 6-mercaptopurine had a recurrence of pyoderma. No patient experienced significant toxicity. CONCLUSION: Intravenous cyclosporine is the treatment of choice for pyoderma gangrenosum refractory to steroids and 6-mercaptopurine should be used as maintenance therapy.
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19/166. Inflammatory bowel disease, azathioprine and skin cancer: case report and literature review.

    A 42-year-old blond Caucasian woman taking azathioprine for 8 years developed an intra-epidermal carcinoma of the shin. She regularly used a sun bed to maintain a tan. Although the increased risk of non-melanoma skin cancer in immunosuppressed transplant recipients is well recognized, patients with Crohn's disease are not currently warned of the risk of exposure to ultraviolet light. Individuals with inflammatory bowel disease who take azathioprine, especially those with a fair complexion, should be informed of the potential dangers of sun bathing and should be advised to limit sun exposure.
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ranking = 3
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20/166. Chronic inflammatory bowel disease in a patient with common variable immunodeficiency.

    common variable immunodeficiency (CVID) is a primary defect of the immune system involving an increased risk of respiratory and digestive tract infections and autoimmune diseases. Recently, it has been reported that chronic inflammatory bowel disease (CIBD) might occur with increased frequency (20%) in patients with CVID. A nine-year-old boy with CVID developed CIBD during follow-up and periodic intravenous immunoglobulin administration. serum tumor necrosis factor-a concentration, which is suggested to show disease activity in CBD, was very high. The patient's radiological evaluation, both in active and remission periods, had characteristic features of CBD. We herewith present and discuss this case with both diseases, CVID and CIBD.
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ranking = 4
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