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Dafne Fernandes Machado,1 Stela Souza Peña,1 Mariana Carneiro Alves,1 Giovana Capecci Siqueira,1 Leonardo Amarante Pereira,1 Samantha Sanches de Carvalho,2 Thais Moura Gascon,2 Beatriz da Costa Aguiar Alves,2 Glaucia Luciano da Veiga,1,2 Fernando Luiz Affonso Fonseca,1– 3 Vagner Loduca Lima,1 Priscilla Ballalai1 1Ophthalmology Discipline, Centro Universitário Saúde ABC/ Faculdade de Medicina Do ABC, Santo André, Brasil; 2Clinical Analysis Laboratory, Centro Universitário Saúde ABC/ Faculdade de Medicina Do ABC, Santo André, Brasil; 3Department of Pharmaceutical Sciences, Universidade Federal de São Paulo, Diadema, BrasilCorrespondence: Dafne Fernandes Machado, Ophthalmology Discipline, Centro Universitário Saúde ABC/ Faculdade de Medicina do ABC, Av. Lauro Gomes, 2.000, Santo André, São Paulo, 09060-870, Brasil, Tel +55-114993-5400, Email dafnefm13@gmail.comAbstract: Choroidal osteoma is a rare, benign, osseous choristoma presenting as an orange-yellow, well-defined fundus mass. It presents unilaterally in most cases, has a predilection for the female sex, and favor a juxtapapillary location, becoming clinically manifest when it involves the macula. Almost 60% of eyes with osteoma may suffer significant visual loss. Choroidal osteomas can easily be mistaken for other conditions with similar presentations. We herein report two rare cases presentation of choroidal osteoma: a 74-year-old male with 2-year blurred vision in the right eye (RE) initially misdiagnosed with age-related macular degeneration received intravitreal injections and after a multimodal evaluation, following cataract surgery in RE, confirmed to be choroidal osteoma and a 19-year-old female with a history of choroidal hemangioma presented with blurred vision in her left eye (LE), with examination revealed an irregular orange-yellow lesion along the superotemporal arcade with serous macular detachment, later diagnosed as choroidal osteoma.Keywords: choroidal osteoma, osseous choristoma, visual loss, case reports