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M.D. Samsun Training and Research Hospital, Eye Clinic, Samsun/TURKEY Two sisters ages 10 and 11 were admitted to our clinic for control examination. Vision level of the case aged 10 (1st case) was 1.0 on both eyes, and corrected vision level of the case aged 11 (2nd case) was 0.6 on both eyes with Snellen scale. Anterior segment examination of both cases was detected naturally; their intraocular pressure was measured normally. During the fundus examination pigmented bone spicules was monitored in retina periphery of both cases, being more intensely in 2nd case. It has been learnt that they had aural deficiency and balance disorder in their detailed anamnesis. Usher syndrome is an autosomal recessive defect associated with hearing loss, balance disorder and retinitis pigmentosa. Eye problems also should be questioned in the child patients admitted with complains of hearing. Keywords : Usher syndrome, retinitis pigmentosa, hearing loss