Isolated sphenoid sinusitis is rather rare. Fungal sinusitis is common in immunocompromised patients. In this case report the authors describe an immunocompetent patient with isolated sphenoid fungal sinusitis with intracranial extension with a review of published literature. Unfavorable location and poor ventilation have been attributed as the probable factors involved in isolated sphenoid sinusitis. Considering the location of sphenoid sinus (close to skull base, optic nerve and great vessels) infections involving this sinus is fraught with dangerous complications.