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Background Inflammatory pseudotumors are ubiquitous lesions characterized by a polymorphous inflammatory infiltrate containing plasma cells and lymphocytes. In the central nervous system, this pathological condition is rare and the association with Crohn's disease has never been described. Case Description A 31-year-old woman with a history of Crohn's disease was referred to our department for progressive headaches and nausea. Neurological examination was normal. Magnetic resonance imaging showed an irregular heterogeneous enhanced mass infiltrating the left cerebellar hemisphere. Total resection was performed and pathological examination led to the conclusion of an inflammatory pseudotumor. Conclusion To our knowledge, this case is the first describing an intra-cerebral inflammatory pseudotumor associated with an inflammatory bowel disease. The diagnosis of an extradigestive location of Crohn's disease was excluded by pathological examination. Although the precise cause of this association remains unknown, it could be hypothesized that the intra-cranial lesion could be the result of the immunosuppressive therapy given for Crohn's disease, or, more likely, could be a part of a systemic dysimmune process.