Osteoma cutis is a rare condition involving the formation of bone in skin or subcutaneous tissue. This may be a primary event or, more often, secondary to an inflammatory, traumatic, or neoplastic process. There is little evidence in the literature of this condition involving the eye or ocular adnexal structures. The authors report two remarkably similar cases involving children with congenital isolated osteoma cutis involving the left lateral canthus. © 2009 American Association for Pediatric Ophthalmology and Strabismus.