A case report of intestinal obstruction caused by cryptogenic multifocal ulcerous stenosing enteritis

Chang, Chun–Ju; Jiang, Chen; Miao, Yaoyao; Fang, Bin; Zhang, Lili

doi:10.1186/s12876-020-01450-5

Cited by 3 publications

(5 citation statements)

References 9 publications

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“…Ulcers of CEAS have been described as shallow oblique, circular, or longitudinal with discrete margins in case series of the endoscopic findings of CEAS[ 13 , 19 ]. CEAS-like diseases without SLCO2A1 mutations have also been reported, including inherited eicosanoid metabolic disorders, inherited human cPLA2α deficiency, and cryptogenic multifocal ulcerous stenosing enteritis; histologically, however, these diseases show nonspecific inflammatory cell infiltration predominantly by neutrophils, rather than eosinophils[ 20 , 21 ]. The present case thus meets the diagnostic criteria for CEAS, but the diagnosis of EoN was reasonable based on the histological findings and the therapeutic course and responsiveness.…”

Section: Discussionmentioning

confidence: 99%

Eosinophilic enteritis requiring differentiation from chronic enteropathy associated with SLCO2A1 gene: A case report

Kimura¹,

Jimbo²,

Arai³

et al. 2023

World J Gastroenterol

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BACKGROUND Eosinophilic gastrointestinal disease (EGID) is a disorder characterized by infiltration of eosinophils causing mucosal damage and dysfunction of the gastrointestinal tract. The endoscopic findings of eosinophilic enteritis (EoN), an EGID variant, are nonspecific and occasionally difficult to diagnose. In contrast, chronic enteropathy associated with SLCO2A1 (CEAS) is a chronic persistent small intestinal disorder characterized by endoscopic findings such as multiple oblique and circular ulcers. CASE SUMMARY We report the case of a 10-year-old boy who had suffered abdominal pain and fatigue for the preceding 6 mo. He was referred to our institute for investigation of suspected gastrointestinal bleeding because of severe anemia with hypoproteinemia and positive fecal human hemoglobin. The upper and lower gastrointestinal endoscopic findings were normal; however, double-balloon small bowel endoscopy showed multiple oblique and circular ulcers with discrete margins and mild constriction of the intestinal lumen in the ileum. The findings were highly consistent with CEAS, but urine prostaglandin metabolites were within normal limits, and no previously reported mutations in the SLCO2A1 gene were identified. Histological evaluation demonstrated moderate to severe eosinophilic infiltration localized to the small intestine suggesting a diagnosis of EoN. Clinical remission was maintained with montelukast and a partial elemental diet, but emergent surgery for bowel obstruction due to small intestinal stenosis was performed two years after the initial treatment. CONCLUSION EoN should be considered in the differential diagnosis of CEAS-like small intestinal ulcerative lesions and normal urinary prostaglandin metabolite levels.

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Section: Discussionmentioning

confidence: 99%

Eosinophilic enteritis requiring differentiation from chronic enteropathy associated with SLCO2A1 gene: A case report

Kimura¹,

Jimbo²,

Arai³

et al. 2023

World J Gastroenterol

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“…Cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is chronic and recurrent stenosis from idiopathic multifocal strictures and ulcerations of small bowel 1 . It is a rare enteropathy with just over 60 cases in literature, often misdiagnosed as Crohn's disease or NSAID‐induced enteropathy 2,3 . The ‘gold standard’ tool for this condition is capsule endoscopy or double balloon enteroscopy 4 .…”

Section: Figmentioning

confidence: 99%

“…In our case, capsule endoscopy was utilized as the primary investigative tool, however diagnosis was confirmed with histology. Due to low incidence, clinicians may have difficulty diagnosing the condition based on endoscopy unless there is a high clinical suspicion 3 . Table 1 demonstrates that although macroscopically similar, there are some distinguishing microscopic characteristics.…”

Section: Figmentioning

confidence: 99%

“…1 It is a rare enteropathy with just over 60 cases in literature, often misdiagnosed as Crohn's disease or NSAID-induced enteropathy. 2,3 The 'gold standard' tool for this condition is capsule endoscopy or double balloon enteroscopy. 4 We present a 59-year-old man with recurrent abdominal cramps and chronic iron deficiency anaemia, diagnosed with CMUSE on histology.…”

mentioning

confidence: 99%

“…Due to low incidence, clinicians may have difficulty diagnosing the condition based on endoscopy unless there is a high clinical suspicion. 3 Table 1 demonstrates that although macroscopically similar, there are some distinguishing microscopic characteristics.…”

mentioning

confidence: 99%

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