Hepatosplenic T-cell lymphoma (HSTCL) is a rare, aggressive malignancy that has been associated with thiopurines during the treatment of inflammatory bowel disease (IBD). We present a case of a 26-year-old man with Crohn's disease on infliximab without prior thiopurine exposure who developed HSTCL. His diagnosis was confirmed through flow cytometry, fluorescence in situ hybridization, and bone marrow biopsy. This is the sixth case of HSTCL in patients with IBD without prior thiopurine exposure and highlights the need for a comprehensive risk-benefit discussion with patients on the various treatment modalities for IBD.