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Inflammatory myofibroblastic tumor of ileum with intussusception in adult. A 62-year-old man was referred to our department with suspected intussusception due to an ileal tumor. Tumor markers including soluble interleukin-2 receptor were not elevated. Contrast-enhanced computed tomography and color Doppler ultrasonography showed a distinct ileal tumor without intratumoral blood flow or surrounding lymphadenopathies. Retrograde single-balloon enteroscopy revealed a submucosal tumor in the ileum that was hard and ulcerated. Partial intestinal resection was performed, and the lesion was diagnosed as an inflammatory myofibroblastic tumor. The patient had no recurrence over 2 years without additional treatment after surgery. This rare tumor should be kept in mind as a cause of ileal intussusception in adult and a multidisciplinary approach is vital to characterize it.