This study aims to describe an atypical presentation of peripapillary retinoschisis (PPRS) in a young myopic patient. A 14-year-old female with high myopia −10.50 diopters in the right and −12.0 diopters in the left eye and good visual acuity (20/20) in both eyes. She presented with splitting of the inner retinal layers in the superior peripapillary quadrant as an incidental finding on spectral-domain optical coherence tomography (SD-OCT) on her left eye. The macula and outer retinal layers were unaffected and it was not associated with any other ocular pathology except myopia in both eyes. Our patient represents an atypical form of PPRS determined incidentally on SD-OCT with schisis of inner retinal layers without macular involvement.Keywords: Myopia, optical coherence tomography; peripapillary retinoschisis; young patient.