We report a case of sinus mucormycosis in a patient receiving infliximab for Crohn’s disease (CD). A 41-year-old white female with a history of gastroesophageal reflux disease, well-controlled diabetes, and ileocecal CD developed right-sided facial pain and high fevers, with computed tomography scan confirming sinusitis. She had been receiving both low-dose azathioprine and scheduled infliximab for her CD. A sinus biopsy was procured endoscopically which grew mucormycosis. All immunosuppressive agents were immediately discontinued, and the patient underwent multiple debridement procedures of the right sinuses. Amphotericin B lipid complex and posaconazole were administered to the patient. Repeat laboratory and imaging study demonstrated clearance of the infection approximately 30 days after diagnosis. The patient’s CD did not flair during withdrawal of immunosuppressive medications, and the patient completed 6 months of posaconazole therapy. Clinicians should be aware of the possible development of this potentially catastrophic infection in patients receiving infliximab, especially if such patients have other risks for mucormycosis, such as diabetes.

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