Cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is a rare disease of unclear origin and pathophysiology. Treatment is symptomatic, with no particular medical therapy except for corticosteroids. We describe a patient with steroid-refractory CMUSE. A 25-year-old woman underwent bowel surgery 4 times because of small-bowel obstruction. She was diagnosed with CMUSE. Despite steroid treatment and immunosuppressive therapy, she continued to complain of recurrent gastrointestinal symptoms and persistent anemia. To avoid short-bowel syndrome in our patient, new medical treatment is necessary for steroid-refractory CMUSE.