Abstract:We present an atypical case of rhino-orbital mucormycosis in an immunocompetent patient and a literature review on this topic. A 17-year-old immunocompetent male presented with 2-month history of painful slowly progressive swelling of the left eye. The diagnosis of mucormycosis was made by histopathological examination of tissue specimens. The patient had successful eradication of the fungal infection with the use of multiple treatment modalities including aggressive endoscopic excision and debridement of ethmoid and maxillary sinuses, left orbital exenteration, intravenous amphotericin B and intraorbital amphotericin B. This case suggests that rhino-orbital mucormycosis can occur in a young healthy individual. Early diagnosis with aggressive medical and surgical management is vital to successfully treat this rare and potentially fatal disease.