Case report of a pair of siblings with cryptogenic multifocal ulcerating stenosing enteritis

Abstract

Rationale: The rare disease cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is characterized by multiple and recurring small intestinal ulcers with stenosis of unknown causes. In clinic, it is difficult to be differentiated from the inflammatory bowel disease, especially the Crohn disease. Patient concerns: Here we report a pair of siblings who suffered from long-time anemia and abdominal pain and misdiagnosed with inflammatory bowel disease (IBD) for many years. Diagnoses: They were finally diagnosed with CMUSE with intestinal obstruction. Interventions and outcomes: They both accepted surgical treatment and recovered well. No abdominal symptom appeared in the two-year follow-up. Lessons: This report underscores that CMUSE patients may have a long course of suffering from anemia and abdominal pain, normal inflammatory markers and normal colon, and sometimes have a family history of CMUSE. Surgery of segmental bowel resection is a good way to solve intractable intestinal obstruction in CMUSE.