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Etlik City Hospital,Eye Clinic,Ankara,Turkiye DOI : 10.37845/ret.vit.2024.33.45 Combined hamartoma of the retina and retinal pigment epithelium (RPE), which was first described in 1973, is a unilateral and extremely rare malformation in which the retina, RPE and adjacent vitreous are effected. In this report, we present a 9-year-old case of Stage 1 combined hamartoma, who had previously been operated on due to strabismus and was referred to us with the preliminary diagnosis of choroidal nevus. The aim here is to highlight the early imaging findings of retina and RPE combined hamartoma, which can cause serious vision loss in childhood and can be confused with intraocular tumors, and to facilitate early diagnosis of the lesion. Keywords : Combined hamartoma of the retina and retinal pigment epithelium, Stage 1, Optical coherence tomography