Background and Purpose: Rhinocerebral mucormycosis is a rare fatal fungal infectionwhich is on a growing trend, particularly among immunocompromised patients.Immunosuppressive drugs, including corticosteroids and antimetabolites, increase therisk of this infection. Herein, we reported the case of fulminant rhinocerebralmucormycosis in a patient with ulcerative colitis receiving azathioprine andcorticosteroid.Case report: A 58-year-old woman was admitted to the hospital in a state of coma withan extensive necrosis in her nose. She was afflicted with intestinal bleeding after 1month of fasting and was treated with azathioprine and a high dose of prednisolone forulcerative colitis 2 months prior to hospital admission. The direct microscopicexamination of the necrotic tissues of the paranasal sinuses showed several non-septatehyphae consistent with Mucorales. Culture media yielded Rhizopus species, which wasidentified as Rhizopus oryzae by internal transcribed spacer polymerase chain reactionsequencing. Despite the implementation of surgical and pharmaceutical (liposomalamphotericin B) treatments, the patient expired after 2 weeks of admission.Conclusion: The gastroenterologists should be aware of the adverse effect ofimmunosuppressive drugs they prescribe for the treatment of inflammatory boweldisease.