Rationale: Inflammatory myofibroblastic tumor (IMT) is an extremely rare soft tissue neoplasm consisting of the proliferation of fibroblastic-myofibroblastic cells with inflammatory infiltrates. It is known to occur in many parts of the body and can generally present with benign or locally recurrent behavior. Anaplastic lymphoma kinase is a specific diagnostic marker for IMT, and approximately 50% of IMT patients have anaplastic lymphoma kinase gene rearrangements. Reports of the female genital tract are rare.Patient concerns: Two patients (a 32-year-old multigravida and a 22-year-old nullipara) visited our clinic because of abnormal uterine bleeding and a uterine mass.Diagnoses: Histopathological examination, immunohistochemical markers, and fluorescence in situ hybridization confirmed the presence of a rare uterine IMT.Interventions: The masses were completely resected via hysteroscopy. The multigravida recurred rapidly in terms of symptoms and images, whereas the nullipara was complaint-free during the follow-up period. Finally, the multigravida underwent hysterectomy and bilateral salpingectomies.Outcomes and lessons: Uterine IMTs can be easily overlooked because of their extremely low incidence rate and insufficient awareness among clinicians; however, uterine IMTs need to be considered in the differential diagnosis of uterine masses. Possible differences in the biological behavior of IMT may exist in different individuals.