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Introduction: To report the unusual fundus features of a case with unilateral Duane retraction syndrome (DRS) with same-side extensive macular retinoschisis. Methods: A 75-year-old woman was diagnosed to have DRS type 3 and several multimodal fundus imaging modalities were performed. Results: There was limited abduction and adduction, globe retraction, and narrowing of the palpebral fissure on the adduction of the left eye without a compensatory face turn. Concurrently, spectral domain optical coherence tomography revealed marked macular retinoschisis and severe vitreoretinal traction without any evidence of dye leakage or pooling on fluorescein angiography in the left eye. Discussion: Various ocular abnormalities may rarely accompany DRS and the present case is the first reported case of most likely coincidental macular retinoschisis in association with DRS.
