The aim of this case report was to present an unusual case of peripapillary retinoschisis (PPRS) associated with ocular hypotony after glaucoma surgery. It refers to a 78-year-old man with primary open-angle glaucoma who developed PPRS while hypotonous. Optical coherence tomography of the peripapillary and the macular area of the right eye revealed PPRS temporally and nasally to the optic disc, more prominent at the level of the outer nuclear layer and less so at the inner nuclear layer. The PPRS completely regressed after 1 month of treatment and restoration of intraocular pressure to normal levels. This case report highlights the fact that PPRS in glaucoma patients may present in the setting of ocular hypotony and appears to resolve when the hypotony is successfully managed. Hydrostatic pressure gradient across retinal vasculature that allows movement of fluid into the extracellular spaces is a potential mechanism for the development of PPRS in ocular hypotony.