| Summary: | Purpose: To describe a new family with benign yellow dot maculopathy. Observations: A young male patient was found to have bilateral multiple small yellow dots in both maculae. Visual acuity and color vision were normal, and no pathological findings were demonstrated on automated visual field, optical coherence tomography (OCT) and electrophysiological testing. Examination of his parents revealed similar findings in his mother, suggesting autosomal dominant inheritance. No deterioration of vision occurred over long term follow up. These findings are consistent with the newly described phenotype of benign yellow dot maculopathy. Conclusions and importance: This is the first report of patients with benign yellow dot maculopathy since its original description, and the first to document it in a family of North African descent. This report will serve to raise awareness to this phenotype, which may be more common than currently known. Keywords: Yellow dot, Maculopathy, Phenotype, Familial
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