Ozanimod is an oral sphingosine-1-phosphate receptor modulator. Although it can be an effective drug for the induction and maintenance of remission in patients with moderately to severely active ulcerative colitis, there have been a few reported cases of various malignancies after exposure to this small molecule. We describe a unique case of biopsy-proven Kaposi sarcoma of the skin and colon in a patient with biologic-resistant ulcerative colitis after treatment with ozanimod for 2 months. Given the potential risk of malignancy associated with this agent, physicians should be aware of this rare adverse event.