Abstract
PURPOSE: To describe a unique case of unilateral benign yellow dot maculopathy. OBSERVATIONS: A 25-year-man was evaluated after incidental finding of yellow dots in the right macula. The findings of examination and multimodal imaging were in keeping with a diagnosis of benign yellow dot maculopathy. CONCLUSIONS AND IMPORTANCE: Benign yellow dot maculopathy is a recently described entity with either a sporadic or dominant inheritance pattern. This is the first known report of the characteristic findings of this phenotype presenting unilaterally.