The authors present a case report of a devastating bilateral optic neuropathy and orbitopathy initiated by a contiguous fungal sinusitis, in an apparently immunocompetent young man. The causative organism, Bipolaris hawaiiensis, and other species classified in the genus Bipolaris, are being recognized with increased frequency as causes of several ophthalmic and systemic disorders in both immunocompromised and normal hosts. A literature review demonstrates the variety of clinical presentations with emphasis on those cases which may present to the ophthalmologist.